the lived experiences of parents of children with
TRANSCRIPT
TheLivedExperiencesofParentsofChildrenwithPolyposisDiagnoses:AdvocatingHealingRelationships
AndreaToo
Thesissubmittedtothe
FacultyofGraduateandPostdoctoralStudies
inpartialfulfillmentoftherequirementsfor
theMaster’sofArtsinEducationinEducationalCounselling
FacultyofEducation
UniversityofOttawa
©AndreaToo,Ottawa,Canada,2016
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
ii
Abstract
Whilemuchresearchhasbeenconductedontheexperiencesofindividualswith
inflammatoryboweldiseases,thereremainsadearthofresearchconductedonthose
affectedbypolyposisconditions.Asaresult,littleisknownaboutthelivedexperiences
ofthosewithpolyposisconditions,especiallyinthecasesofparentsofpediatricpatients
withtheseconditions.Thisstudyqualitativelyexploredthelivedexperiencesofparents
ofchildrenwithpolyposisconditions,specificallyJuvenilePolyposisSyndromeand
Peutz-JeghersSyndrome.Hermeneuticphenomenologywasusedtoexplorethelived
experiencesofsevenparentsofchildrendiagnosedwithpolyposisconditionsthrough
semi-structuredinterviews.CollecteddatawasanalysedusingLindsethandNorberg
(2004)’sPhenomenologicalHermeneuticalMethodforResearchingLivedExperience.
Intotal,fourmajorthemescomprisingoftwelvesub-themeswererevealed.
Parentsdiscussedfeelinggratefulfortheuseoffamily-centredapproachesbytheir
children’sphysiciansaswellasaccesstomedicalcarefortheirchildren,which
encouragedthemtodemonstrateaproactiveapproachtowardstheirchildren’shealth
maintenance.Furthermore,theyexplainedthatwhileseekinginformationconcerning
theirchildren’sconditionswasanxiety-inducing,discussingtheirexperienceswith
otherswithsituationssimilartotheirswasvalidatingandinformative.Theparticipants
describedtheimportanceofadvocatingfortheirchildrenwithinandoutsideofthe
medicalsystem,andtheresponsibilitytheyfeelinteachingtheirchildrentoundertake
theadvocatingprocessforthemselves.Lastly,theparentsreflectedontheimpacttheir
children’sdiagnoseshavehadontheirrelationshipswiththemselves,theirfamiliesand
theirsupportnetworks.Overall,thefindingsfromthisstudyarein-linewithfindings
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
iii
frompriorresearch,exceptinthecaseofthe‘Teachingthechildrentospeakfor
themselves’themewhichprovestobeanovelcontributiontotheliterature.
Thesharedkeyaspectsofthephenomenonindicatethatfocusshouldbeplaced
ontheutilizationoffamily-centredcarebyphysicians,thedevelopmentofsupport
groupsforparents,andoneducatingphysiciansonhowtobestfacilitateparentsasthey
modeladvocatingbehaviourstopediatricpatients.Thisstudyprovidesinsightintothe
livedexperiencesofparentsofchildrenwithpolyposissyndromes,informingthe
medicalcommunityofhowtheneedsofthisgroupcanbebettermet.Furthermore,the
qualitativenatureofthisresearchwillprovidethepolyposis,chronicillnessandrare
illnessliteratureswithinformationithasbeenlacking,usingavaluablemethodological
perspective.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
iv
Acknowledgements
AsIreflectonthepastfewyearsofthedevelopmentofthisthesis,Iamfilledwith
immeasurablegratitudeandalittlebitofshockthatthischapterofmyjourneyisfinally
complete.Therearemanypeoplewithoutwhomthisprojectwouldnothavebeen
successful.IwouldliketoexpressmygratitudetomyresearchsupervisorDr.André
Samsonforhissupervisionandencouragementthroughoutthislongprocess.Thankyou
aswelltomythesiscommittee,Dr.JaniceBarkey,Dr.EvaTomiakandDr.Katherine
Moreau,whoseoptimismandbeliefintheresearchprocessprovedtobecontagious,
eveninthemostwantingoftimes.Thankyoutomyfriendsfortheirsupportduringthe
innumerablerantsessions,coffeedates,longdistancephonecallsandcelebrationsof
eventhetiniestofsuccesses–Iamsofortunatetobeabletogrowalongsideofyou.
ThankyoutoTim,whobroughthumourtothisprocess.Laughingwithyoucontinuesto
bemyhappyplace.Thankyoutomyparentsandsisterforsimplybeingthebest.Mom,
Dad,andKristen,thankyouforhelpingmetofindwithinmyselfastrengthIdidn’tknow
Ihad.Iamforevergrateful.
Andlastbutnotcertainlynotleast,thankyoutothemenandwomenwhoso
willinglyandbravelysharedtheirstories.Thisprojectwouldnothavebeenpossible
withoutyou.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
v
TABLEOFCONTENTSAbstract iiAcknowledgements ivTableofContents vListofAppendices viListofTables viListofFigures viCHAPTER1:INTRODUCTION 1DescriptionoftheResearchProblem 1RelevanceofthisResearch 2OutlineoftheThesis 3CHAPTER2:LITERATUREREVIEW 4Peutz-JeghersSyndrome 4JuvenilePolyposis 5Family-CenteredCare 6RareDiseases 7IncreasedRiskofCancer 10FearofCancer 12Parents’ManagementofChronicIllnessinChildren 15PsychologicalDistress 15RiskandProtectiveFactors 16ParentalCopingStyles 16ExperienceofHope 19FamilyDynamics 19ExperiencesofPolyposisSyndromes 20ObjectivesandResearchQuestions 23ContributionstoKnowledgeandPractice 24CHAPTER3:METHODOLOGY 25PhilosophicalFoundations:HermeneuticalPhenomenology 25RationalefortheUseoftheHermeneuticalPhenomenology 26ResearcherPosition 28Participants 29EligibilityCriteria 29RecruitmentProcess 29InstrumentDevelopment 30DevelopmentoftheInterviewProtocol 30DevelopmentoftheDemographicQuestionnaire 31Demographics 32DataCollection 33DataAnalysis 35MethodologicalRigour 36
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
vi
CHAPTER4:FINDINGS 39UseofIdentifiers 39DescriptionofEssences 39Buildinghealingrelationshipswithinthemedicalcommunity 40
Comingtogetherforthewellbeingofthechild 41Alwaysonthelookout:Doingeverythingpossibletomaintainhealth 46Feelinggrateful:Itcouldbeworse 50
Peekingthroughcoveredeyesatillnessliterature 52Seekingvalidationinsharedexperiences 52
Tuningoutanxiety-inducinginformation 55Perserveringuntilneedsaremet 62
Teachingchildrentospeakforthemselves 62Trustingmaternalintuition 65Takingitonedayatatimebecausethefutureisuncertain 66
Creatinganew‘normal’aseverythingbecomesunsettled 71 Becomingadifferenttypeofparent 71
Trustingtheywillspeaksotheycanbeheard 72Puttingfamiliestothetest:copingwiththeillnessasafamily 74Feelingasthoughnooneunderstands 78
DescriptiveIdentificationofthePhenomenonStructure 84 CHAPTER5:DISCUSSION 86SummaryofFindings 86DiscussionofFindings 87ImplicationsfortheMedicalField 99ImplicationsfortheCounsellingDomain 100Limitations 101AvenuesforFutureResearch 102Conclusions 104REFERENCES 105APPENDICES 125 AppendixA:DemographicQuestionnaire 125 AppendixB:PermissiontoContactParticipantsForm:CoverLetter 127 AppendixC:PermissiontoContactParticipantsForm:StudyDescription129 AppendixD:ParticipantInformationandConsentForm 134 AppendixE:InterviewProtocol 140
LISTOFTABLESTable1:Participants’Demographics 33LISTOFFIGURESFigure1:Buildinghealingrelationshipswiththemedicalcommunitytheme
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
vii
andsub-themes. 40Figure2.Peekingthroughcoveredeyesatillnessthemeandsub-themes. 52Figure3.Perseveringuntilneedsaremetthemeandsub-themes. 61Figure4.Creatinganew‘normal’aseverythingbecomesunsettledtheme andsub-themes 70
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
1
Chapter1:Introduction
DescriptionoftheResearchProblem Colorectalcanceristhethirdmostcommoncancerinwomenandthefourthmost
commoncancerinmenworldwide(Parkin,Bray,Ferlay,&Pisani,2005).Colorectal
cancercanbeattributedtoindividuals’geneticsinapproximately35%ofdiagnoses
(Patel&Ahnen,2012).Polyposissyndromesconferasignificantriskofcancerinthose
diagnosedwithsuchconditions,aspolypsdevelopinginthecoloncanbecomemalignant
(Giardiello,etal.,2000).Theseconditionsareoftendiagnosedatayoungage,prompted
byeitherclinicalsymptomsorgenetictestingbasedonfamilyhistory(Ellis,2004).
Althoughpolyposissyndromeshavebeenstudiedextensivelyintheliterature
fromabiologicalperspective,researchontheexperiencesofindividualsandtheir
familieslivingwiththeconditionsislacking.Researchexploringthepsychosocial
perspectivesofindividualslivingwithgastrointestinalconditions,otherthanpolyposis
conditions,indicatethataffectedindividualsexperiencesocialisolation,and
psychologicaldistress(Casati,Toner,deRooy,Drossman,&Maunder,2000;Graff,
Walker,&Berstein,2009).Moreover,virtuallynoresearchexistsontheexperiencesof
parentsraisingchildrenwithpolyposisconditions.(Theterm‘parent’willhereafterbe
usedtorefertoparents,caregivers,andlegalguardians).Aschildrenrepresentan
inherentlyvulnerableportionofthepopulation,theresponsibilityofcaringfor
polyposis-diagnosedchildrenlaysinparents’hands.Withthehealthpsychology
literaturedemonstratingevidencethatparentsofchronicallyillchildrensuffer
pronouncedresponsestotheirchildren’sconditions,namelypsychologicaldistress,itis
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
2
likelythatthediagnosisofachildwithapolyposissyndromeconfersvulnerabilityupon
bothpediatricpatientsandtheirparents(Kazaketal.,2005).
Uniquetopolyposisconditionsaretheconditions’heightenedlikelihoodofcancer
development.Researchindicatesthatindividualswhohaveanincreasedriskof
developingcolorectalcanceroftendonotcomplywithscreeningrecommendationsdue
totheirfearofcancer(Bleiker,etal.,2005).Itisofparamountimportancethatthe
experiencesofparentsofchildrenwithpolyposissyndromesbeexploredinordertogain
insightintothewaysinwhichtheycopeandadapttotheirchildren’sillnessesandto
betterunderstandtheeffectssuchexperienceshaveonthemedicaldecisionstheymake
fortheirchildren.Explorationwouldalsogiveusbetterinsightintotheneedsofthese
parentsandhowsuchneedscanbemet.
Assuch,thisstudyseekstoexplorethelivedexperiencesofparentsofchildren
withpolyposissyndromes.Thisthesiswillfocusontwopolyposissyndromes,Peutz-
JeghersSyndromeandJuvenilePolyposisSyndrome.Thequalitativeapproachguiding
thisstudycomplementsthepredominantlyquantitativedatathatcurrentlydominates
theresearchliterature.Thefindingsrevealedwithinthisstudyareframedinawaythat
canbetranslatedtomoreappropriatesupportforparents,andsubsequently,bettercare
fortheirchildren.Furthermore,thisresearchwillhelptofillthegapsintheliterature
pertainingtotheexperiencesofparentsofchildrenwithpolyposisconditions,andona
moregenerallevel,chronicandrareconditions.
RelevanceofthisResearch Theexplorationofthisunder-investigatedtopiccontributestoboththeresearch
literatureandpracticalworkofclinicians.Thecomplexnatureofthistopicindicatesthat
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
3
itsfindingswillberelevanttoseveraldifferentresearchdomainsincludingtherare
illness,chronicillness,andparentalexperiencesliterature.Thequalitativeapproachof
theprojectwillalsoprovidetheliteraturewithuniqueexperientialinsights,whichare
currentlyunderrepresented.Practicallyspeaking,itisimportanttounderstandthe
experiencesofparentsofchildrenwithpolyposisconditionsinordertomoreeffectively
servetheirpsychosocialneedsandtobetterfacilitatethemedicalcareoftheirchildren.
OutlineoftheThesis Thisstudyisdividedintofivechapters.Followingtheintroductorychapter,in
Chapter2Isummarizetheresearchliteraturethatinformsthecurrentstudyanddiscuss
theobjectivesandresearchquestionsguidingtheproject.InChapter3,Iexplainthe
hermeneuticalphenomenologymethodologyandmyrationaleforitsuse.Ialsodiscuss
howthedatacollectionanddataanalyseswereconductedandthemeansbywhich
methodologicalrigourwasensured.InChapter4Idescribethefindingsrevealedinthe
dataanalysisprocessusingparticipantquotationsandexplaintherelationsbetweenthe
themes.Inthefinalchapter,Chapter5,Ipositionthefindingswithintheresearch
literature,discusstheimplicationsofthefindings,andthefinalconclusionsthatcanbe
drawnfromthestudy.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
4
Chapter2:LiteratureReview
Peutz-JeghersSyndrome
Peutz-JeghersSyndromeisanautosomal-dominantconditioncausedbya
mutationoftheSTK11gene(Chow,Meldrum,Crooks,Macrae,Spigelman,&Scott,2006).
ApproximatelyonequarterofPeutz-JeghersSyndromecasespresentasdenovo,wherein
themutationpresentsitselfforthefirsttimeintheindividual(Schreibman,Baker,Amos,
&McGarrity,2005).Theconditionaffects1in50,000to1in200,000livebirths(Burt,
2002;Utsunomiya,Gocho,Miyanaga,Hamaguchi,Kashimuri,Aoki,&Komatsu,1975).
Peutz-JeghersSyndrome’smostcommonclinicalsymptomsincluderectalbleeding,pain
intheabdomenandanemiaaswellaspotentiallyfatalobstructionandextrusionofthe
polyps(Utsonomiyaetal.,1975).IndividualsdiagnosedwithPeutz-JeghersSyndrome
generallymeetthediagnosticcriteriaproposedbyGiardielloandcolleagues(1987).
Patientsmusthavepathologicevidenceforhamartomatousintestinalpolypsandmeet
twoofthefollowingrequirements:polyposisofthesmallintestine,afamilyhistoryof
Peutz-JeghersSyndrome,anddiscolouredmaculesontheoralmembrane,lips,fingers,
feetandgenitals.AdiagnosisofPeutz-JeghersSyndromepredisposesindividualstoa
higherriskofcancer(Giardielloetal.,1987;Spigelman,Murday,Phillips,1989;vanLier
etal.,2011).
Itisrecommendedthatphysiciansscreenat-riskindividualsfrombirth,checking
yearlyforsymptomsofPeutz-JeghersSyndrome(Giardiello&Trimbath,2006).Because
30%ofPeutz-JeghersSyndromepatientsexperiencebowelobstructionbefore10years
ofage,itisalsoadvisedthatgenetictestingbeofferedtoat-riskchildrenat8yearsof
age,eveniftheyareasymptomatic(Hinds,Philp,Hyer,&Fell,2004).
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
5
JuvenilePolyposis
JuvenilePolyposisisaconditionthataffects1in160,000individuals(Chevrel&
Gueraud,1975).InthemajorityofJuvenilePolyposisSyndromecases,75%,presentwith
afamilyhistoryofthecondition,while25%ofcasespresentasdenovo(Schreibman,
Baker,Amos,&McGarrity,2005).Theterm‘juvenile’inthecondition’snameisusedto
characterizethetypeofpolypfoundinthecolonratherthantheageofonsetofthe
condition.However,clinicalsymptomsgenerallybegintopresentthemselvesbefore10
yearsofage(Ellis,2004).IndividualswithJuvenilePolyposisexperiencechronic
bleedinginthegastrointestinaltract,anemia,diarrhea,prolapsedpolyps,andabdominal
crampsandpain(Merg&Howe,2004).InordertobediagnosedwithJuvenilePolyposis
Syndrome,theindividualmustmeetthefollowingdiagnosticcriteria:Eliminationofthe
possibilityofCowdenSyndromeorBannayanRileyRuvalcabaSyndromediagnoses,
presentwithmorethan5juvenilepolypsinthegastrointestinaltractoranynumberof
confirmedjuvenilepolypswithafamilyhistoryofthecondition(Jass,Williams,Bussey,
&Morson,1988).AswithPeutz-JeghersSyndrome,adiagnosisofJuvenilePolyposisalso
confersagreaterriskofdevelopingmalignancies(Chow&McCrae,2005).Expertsinthe
gastroenterologyfieldadviseindividualswithJuvenilePolyposisdiagnosestoparticipate
inregularbloodexaminationsandendoscopies,withremovaloffoundpolyps(Dunlop,
2002).ItrecommendedthatindividualswithJuvenilePolyposisSyndromeundergo
routinemedicalsurveillancepracticessuchasendoscopy,everyonetotwoyears,
althoughthisisdependentonthelocationandseverityofthepolyps(Dunlop,2002).
IndividualsdiagnosedwithJuvenilePolyposisSyndromeshouldalsoparticipatein
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
6
geneticcounselingandtesting,withrelevantinformationtobesharedwithat-riskfamily
members(Chow&McCrae,2005).
Family-CenteredCare
Family-centeredcareisanapproachcomprisingofapartnershipbetween
medicalpractitionersandfamiliesinthemedicaldecision-makingandmedicalcare
process(Kuo,Houtrow,Arango,Kuhlthau,Simmons&Neff,2012).Althoughconsidered
tobethepediatricstandardofcarewithinthemedicalcommunity,family-centeredcare
isofteninadequatelyimplemented(Kuoetal.,2012).Familycenteredcarecomprisesof
severalelements:understandingtheimportanceofthefamilyinthechild’slife,
respectingthediverseethnic,religiousandsocio-economicbackgroundsoffamilies,
acknowledgingstrengthswithinfamiliesanddifferentcopingmethods;encouraging
support-seekingbehaviours;integratingdevelopmentaladaptationstoclinicalpractices,
andengaginginmedicalpracticesthatmeetfamilyneedswithflexibility(Trivette,1993;
Johnson1990;Shields,Pratt,Davis,&Hunter,2007).
Considerableevidenceexistsdemonstratingtheadvantagesofclinicians’useofa
family-centeredapproach.Researchindicatesthatfamilycenteredtherapyisrelatedto
moreeffectivecommunicationbetweenphysiciansandfamilies,improvedmentaland
physicalhealthamongpediatricpatientsandincreasedengagementinmedical
surveillancepractices(Clarketal.,2000;Bonneretal.,2002;Kellyetal.,2000).Thereis
adearthofevidence,however,exploringthefamily-centeredexperiencesofpediatric
patientssufferingfromrareillnessandtheirfamilies.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
7
RareDiseases
Rarediseasesareconditionswithlowprevalencerates,manifestingthemselvesin
fewerthan1in2000individualsbyEuropeanstandards,andfewerthan1in20,000by
Americanstandards(Zurynski,Frith,Leonard,&Elliott,2008).AsPeutz-Jeghers
SyndromeandJuvenilePolyposisSyndromehaveprevalenceratesof1in50,000and1
in100,000respectively,bothoftheseconditionsareconsideredtoberarediseases
(Chevrel&Gueraud,1975;Woo,Sadana,Mauger,Baker,Berk,&McGarrity,2009).Over
8000rarediseaseshavebeenidentifiedworldwide,creatingagreaterimpactthantheir
categorizationwouldimply(Zurynski,Frith,Leonard,&Elliott,2008).Intotal,rare
diseasesareexperiencedby6-10%oftheworldpopulation,affecting30million
Europeansand25millionAmericans(Knight&Senior,2006).
Althoughrarediseasesareexperiencedbyfewcomparedtomorecommon
diseases,theirrarestatusbeliestheirpsychosocial,physicalandeconomicimpacton
thoseaffected(Zurynski,Frith,Leonard,&Elliott,2008).Researchintotheexperiences
ofindividualswithrarediseasesisconsideredtobeanewlyemergingfield,with
researchersfacingmanybarriersinrecruitingstudyparticipantsfromsuchsmallsample
pools(Ettore,2006).Withoutrepresentationoftheexperiencesofindividualswithrare
diseases,theillnessliteratureisbiasedandunrepresentative.
Asparentsofchildrenwithrarediseasesplayanimportantroleascaretakers,
theytooexperiencedifficultiesduringthecaretakingandparentingprocess(Dellve,
Samuelsson,Tallborn,Fasth,&Hallberg,2006).Parentsofchildrenwithrareillnesses
experienceheightenedpsychologicalstresscausedbybarrierstoappropriatemedical
care,suchaslackofmedicalexpertise,diagnosticdelay,uncertaintyoffuturehealth,and
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
8
barrierstonecessarymedicalcare(LeCam,2007;Zurynski,Frith,Leonard,&Elliott,
2008).Parentsofchildrenwithrarechronicillnessesalsoservedifferentroleswithin
thefamilythanparentsofhealthychildren.Theymustexpendextraenergytoward
copingwiththeirchildren’sailments,accepttherealityofthecondition,meettheir
children’smedicalandpsychosocialneeds,advocatefortheirchildren,educate
individualsinthechildren’slives,andcreateasupportteamforthechildren(Dellve,
Samuelsson,Tallborn,Fasth,&Hallberg,2006).Inparticular,mothersareatan
increasedriskofexperiencingpsychosocialdistressrelatedtotheirchildren’sillnesses
comparedtothechildren’sfathers,ormotherswithhealthychildren(Dellve,
Samuelsson,Tallborn,Fasth,&Hallberg,2006).Researchindicatesthatbothmothers
andfathersofchildrenwithrarediseasesgenerallyengageinactivecopingstyles,
activelyseekingoutresourcestobetterunderstandhowtomeettheirchildren’sneeds
(Dellve,Samuelsson,Tallborn,Fasth,&Hallberg,2006).Interestingly,Budych,Helmsand
Schultz(2002)havefoundthatparentsofpediatricpatientsaremorelikelytoengagein
patient-directedinteraction,whereintheparentsofpatientstaketheleadininforming
thephysicianabouttheirchild’scondition,symptoms,thanadultpatientsarelikelyto
advocateforthemselves.Insum,evidencedemonstratesthatparentsofchildrenwith
rarediseasesfeelthattheyareunder-supportedbythemedicalandresearch
communitiesandexperiencesignificantstressesincaringandadvocatingfortheir
childrenwithrareconditions(Zurynski,Frith,Leonard,&Elliott,2008).
Patientsaffectedbyararediseasehavesignificantlydifferentmedical
experiencesthantheircounterpartswithmorecommonconditions.Individualswith
rarediseasescontradictthegeneralphysician-patientinteractionmodelwherebythe
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
9
physicianservesasanexpertonthepatient’sailmentandthepatientactivelyreceives
thephysician’sexpertknowledge(Budych,Helms,Schultz,2012).Incasesofrare
disease,physiciansoftenlacktheexpertiseandexperiencenecessarytohelpthepatient,
possiblyprovidingpatientswithinconsistentinformation(Budych,Helms,&Schultz
2012).Assuch,patientsbecomeexpertsontheirdiagnosesandtheirownbiggest
advocates(Aymé,Kole,&Groft,2008;Budych,Helms,&Schultz,2012).Sucha
phenomenonisdemonstratedbypatients’empowermentasfuellingtheirsearchfor
informationandparticipationinpatientsupportgroups(Aymé,Kole,&Groft,2008).
Positioningpatientsasmedicalexpertsalongsidetheirchildren’sphysicianschangesthe
physician-patientdynamicbyalteringtherolesofthetwoparties(Budych,Helms,&
Schultz,2012).Researchindicatesindividualsvaluetheirprofessionalidentitiesandare
resistanttoalteringtheirviewsofthemselvesinrelationtotheirwork(Chreim,
Williams,&Hinings,2007).Becauseindividualswithrarediseasesandtheirfamilies
oftenareexpertsontheconditions,theyaremorelikelytoconfrontphysicianswhen
theyfeeltheirortheirchildren’smedicalneedsarenotbeingmet.Whentheyfeeltheir
competenceisbeingchallenged,physicianssometimesattempttoregaincontrolofthe
dynamicbyunilaterallymakingdecisionsforthepatientsandtheirfamilies,without
takingthepatients’orfamilies’stancesintoaccount(Budych,Helms,&Schultz,2012).
Preliminaryresearchhasalsobeguntoexplorethepsychosocialexperiencesof
patientswithrarediseasesversustheexperiencesoftheircounterpartswithmore
commondiseases.ResearchbyNispen,vanRijkenandHeijmans(2003)indicatesthat
individualswithrarechronicillnessesexperiencelowerphysicalandpsychosocial
qualityoflifethanpatientswithmorecommonchronicconditions.Theseindividuals
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
10
alsodemonstratedagreaterneedandwantforsocialsupport,eitherthroughother
patientsofrarediseases,clinicians,orphysicians(Nispen,vanRijken,&Heijmans,
2003).
IncreasedRiskofCancer
Parentsofchildrenwithpolyposisconditionsmustnotonlycontendwiththe
rarityoftheconditionsbutmustalsocopewiththerealitythattheirchildren’s
conditionsconferanincreasedriskofthedevelopmentofcancer(Boardmanetal.,1998;
Jass,Williams,Bussey,&Morson,1988).EvidencedemonstratesPeutz-Jeghers
Syndrometobecloselyrelatedtothedevelopmentofbothgastrointestinalandnon-
gastrointestinalcancers,whileJuvenilePolyposisSyndromeismoststronglyassociated
withthedevelopmentofcolorectalcancer(Giardiello,etal.,2000;Jass,Williams,Bussey,
&Morson,1988).
Peutz-JeghersSyndromeconfersariskforcancerthatis18timesgreaterthan
thatofthehealthypopulation(Giardielloetal.,1987).Inameta-analysisbyGiardiello
andcolleagues(2000),theresearchersconcludedthatPeutz-Jegherspatientshavea
cumulativeriskofdevelopingcancerat93%betweentheagesof15and64yearsofage.
Peutz-JeghersSyndromeismoststronglylinkedtothedevelopmentofgastrointestinal
cancer,althoughcasesofbreast,ovarian,cervical,uterine,thyroid,prostateandlung
cancerhavealsobeendocumentedintheresearchliterature(Boardman,etal.,1998).In
particular,womenaremorevulnerabletodevelopingcolorectalcancerasaresultof
Peutz-JeghersSyndromethanmenwiththecondition(Boardman,etal.,1998).
Furthermore,theriskconferredbyPeutz-JeghersSyndromeonwomentodevelopbreast
canceriscomparabletotheriskconferredbyhereditarybreastandovariancancer-
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
11
relatedBRCA1andBRCA2germlinemutations(Giardiello,etal.,2000).
ResearchintotheroleofJuvenilePolyposisSyndromeinthedevelopmentof
cancerhasbeenlessextensivethanthatofPeutz-JeghersSyndrome.Ratesinthe
developmentofcancersinJuvenilePolyposisSyndromecasesvarygreatly,withresearch
demonstratingarangeof17%to55%ofdiagnosedpatientsdevelopinggastrointestinal
cancers.Whilethediscrepancybetweenthetwostudies’riskofmalignancyevaluations
ispossiblybeduetothegeneticnatureoftheJuvenilePolyposisSyndromes-Coburn
andcolleagues(1995)includedbothfamilialanddenovoformsofJuvenilePolyposis
whileHoweandcolleagues(1998)exclusivelystudiedparticipantswithfamilialJuvenile
Polyposis-onecannotdiscounttheroleresearchmethodologyplaysinrevealingresults.
Factorssuchassamplesize,andthepopulationscomprisingthesamples(clinicalvs.
population-based)forexample,differintheaforementionedstudiesandlikelyplaya
roleintheresultsachieved.Atthistime,theliteraturedemonstratesalargerangeof
reportedpenetranceforgastrointestinalcancersfrompolyposisconditions.
AsmalignancyisaverylikelydevelopmentinthePeutz-JeghersSyndromeand
JuvenilePolyposisSyndromeconditions,physiciansadvisepatientstoactivelyengagein
diseasemanagementprograms,althoughrecommendationsvarybyprofessional
(Syngal,Brand,Church,Giardiello,Hampel,&Burt,2015).Physicianswillgenerally
recommendthatindividualsatriskforPeutz-JeghersSyndrome,suchasthosewith
diagnosedimmediaterelatives,beevaluatedyearlyforcommonPeutz-Jeghers
symptomsfrombirth(Giardiello&Trimbath,2006).Childrenwhoareasymptomaticby
theageof8shouldstillbegeneticallytestedforgenesassociatedwithPeutz-Jeghers
Syndrome(Giardiello&Trimbath,2006).Asdiscussed,diagnosedindividualswillbe
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
12
advisedtoundergocolonoscopy,endoscopyyearly.Moreover,gastroenterologistswill
advisethatpolypslargerthan1centimeterberemovedbypolypectomyorenterectomy
(Giardiello&Trimbath,2006).Asameanstomanagingcancerrisk,individualswith
JuvenilePolyposisSyndromeareadvisedtoundergogastrointestinalcolonoscopyand
endoscopyoncesymptomsdevelop,andeveryonetotwoyearsthereafter(Howe,
Mitros,&Summers,1998).Polypsareremovedsurgically,withlargegroupsofpolyps
removedthroughcolectomy(Lindor,McMaster,Lindor,&Greene,2008).Genetictesting
forJuvenilePolyposisSyndromeseekstoevaluatepathogenicvariationsoftheBMPR1A
andSMAD4genes(Haidle&Howe,2015).Consultationwithageneticcounselloris
recommendedtoat-riskandaffectedindividualsinordertobetterunderstandthe
inheritanceandnatureofthecondition.Inadditiontoclosesurveillanceofthecolonfor
malignancies,itisalsoadvisedthatphysiciansbegintoscreenyoungadultpatientsfor
breast,ovarian,testicular,andpancreaticcancers(Syngal,Brand,Church,Giardiello,
Hempel,&Burt,2015).
FearofCancer
Formanyoftheindividualslivingthepolyposisexperience,canceris
“synonymouswithdeath”(Ivanovich&Whelan,1997,204).Fortheparentsofchildren
withincreasedlikelihoodtowardthedevelopmentofmalignancy,afearofsucha
developmenthasbeendemonstratedintheliterature(Aronson,2009).Inadultpatients,
theseattitudesimpactindividuals’likelinesstoengageinillnessmanagement
behaviourssuchassurveillancebehavioursandgenetictesting.Thedecisiontoengage
insurveillancebehaviourscanbepotentiallylifesaving,ascolorectalcancercanbe
effectivelymanagediftreatedearlyenoughinitsdevelopment(Lieberman,1994).
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
13
Whilesuchbeliefsandattitudesconcerningthedevelopmentofcancerstronglyimpact
diagnosedadultindividuals’experiencesofillnesswhilealsoimpactingtheirfamilial
relationships,theimpactofhowsuchattitudesheldbyparentsofpediatricpatients
impactbothparentandchildexperienceshasnotbeenascloselyexplored.
Physicianswilloftenadviseparentsofchildrenatriskforapolyposissyndrome
toundergogenetictesting(Giardiello&Trimbath,2006).Genetictestingisanimportant
toolinanat-riskindividual’ssurveillance,diagnosisanddevelopmentoftreatment
protocol(MacDonald&Lessick,2000).However,genetictestinginchildrenisamuch
morecomplicatedprocessasissuesofconsent,familydynamics,thechild’sabilityto
comprehendandcopewiththeresultspresentthemselves(Tischkowitz&Rosser,2004).
Genetictestingcanincitebothpositiveandnegativereactionsfromindividuals,
dependingontheirresults,copingstylesandexpectations(Shiloh,Koehly,Jenskins,
Martin,&Hadley,2008).
Parentsmustbemindfulthatpre-symptomaticgenetictestingoftheirchildren
mayinterferewithnormalfamilyinteraction,changingthewaychildrenandsiblingsare
treated(Tischkowitz&Rosser,2004).Ifachildisfoundtobeacarrierofcancer-related
gene,parentsmayexperienceguiltfortransmittingthegene.Moreover,non-carrier
siblingsofthecarrierchildmayexperiencesurvivor’sguilt.Parentsmustalsobe
preparedtoaidtheirchildrentocopewiththefeelingsofanxietyaboutfuturehealthand
evolvingidentity(Wertz,Fanos,&Reilly,1994).
Parentsoftendecidewhetherornottohavetheirchildundergogenetictesting
whentheirchildrenarenotyetabletoconsenttotheprocess.Iftheychooseto
participateintheprocess,parentsmustweighthecostsandbenefitsofthetesting.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
14
Furthermore,theymustdecidewhentheywillinformthechildoftheresultsand
whetherfamilymembersandfriendsshouldbenotifiedonbehalfofthechild
(Tischkowitz&Rosser,2004).
Inastudyassessingtheexperiencesof67first-degreefamilymembersof
individualswithcoloncancer,Bastaniandcolleagues(2001)foundthatonly19%of
participantscorrectlybelievedthemselvestohaveanincreasedvulnerabilityof
developingthemalignancy.Theresearchersalsodeterminedparticipants’‘fearoffinding
cancer’tobethegreatestbarrierinpreventinghigh-riskindividualsofengagingin
screeningmeasures,suchascolonoscopyorgenetictesting(Bastani,Gallardo,Maxwell,
2001).ThesefindingsareconsistentwithBleikerandcolleagues’(2005)resultsinwhich
oneofthemainreportedreasonsfornotcomplyingwithscreeningrecommendations
wasfearofthepositivecolorectalcancerresults.Researchassessingtheexperiencesof
individualswithafamilyhistoryofHereditaryNonpolyposisColorectalCancerfound
thatmorethanhalfofparticipantsdeclinedtheopportunitytoundergogenetic
screening(Lerman,etal.,1999).Aronson(2009)suggeststhathigh-riskindividualswho
activelychoosenottoengageinscreeningbehavioursmayrefrainfromdoingsoasan
avoidancecopingmechanismcausedbytheanxietyandstressofdevelopingcancer.
Thesefindingsrevealtousthatpolyposispatients’fearofcancerisprominentenoughto
inhibittheuseofgenetictestingandpossiblylife-savingtreatments.Aswillbediscussed
below,parentsaretheadvocatesanddecision-makersfortheirchildren’smedicalcare,
andsuchafearofcancermayplayaroleintothemedicaldecisionsmade.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
15
Parents’ManagementofChronicIllnessinChildren
Thereexistsanextensiveliteratureontheexperienceofparentingchildrenwith
chronicillness.Researchindicatesthatparentsandthefamiliesofchildrenwithchronic
illnessesaregreatlyimpactedbytheirchildren’sillnessesfromtheinitialdiagnosis
throughongoingtreatment(Tomiak,Samson,Miles,Choquette,Chakraborty,&Jacob,
2007).
Psychologicaldistress.AsbothPeutz-JeghersSyndromeandJuvenilePolyposis
Syndromeconferahighriskofcancer,thefindingsrelatedtoparentalcopingwith
children’scancerexperiencesarehighlyrelevant.Thislineofresearchwillbeusedto
shedlightontheexperiencesofparentalcopingwithchildren’schronicillness,as
researchontheparentingofchildrenwithpolyposissyndromesisvirtuallynon-existent.
Parentsaidingtheirchildrenthroughthecancerexperienceareatincreasedrisk
ofdevelopinganxiety,depressionandevenpost-traumaticstresssymptoms(Dalquist,
Czyzewski,&Jones,1996;Kazak,Boeving,Alderfer,Hwang,&Reilly,2005;Sawyer,
Antoniou,Toogood,Rice,&Baghurst,1993).Specifically,researchdemonstratesthat
mothersandfathersdifferintheirriskfactorsformaladaptation.Theheightofsuch
distressisexperiencedatthetimeofthecancerdiagnosisofthechild,withcontinued
parentaldistressthroughouttheillness(Kazaketal.,2005;Magni,Carli,DeLeo,Tshilolo,
&Zanesco,1983).Researchfindingsareunclearastowhethersuchintenseemotional
reactionsdissipateovertimeorwhetherthelevelofdistresspresentatthetimeof
diagnosispersiststhroughoutthetreatmentregime(Dalquistetal,1996;Kazaketal.,
2005;Sloper,2000).
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
16
Riskandprotectivefactors.Dockerty,Williams,McGee,andSkegg(2000)argue
thatparentsofchildrenwithchronicillnessdonotcompriseahomogenousgroup.
Therefore,riskandprotectivefactorsshouldbetakenintoconsideration.Accordingto
Sloper(2000),motherswhoanticipatedtheexperienceofparentingachildwitha
chronicillnesstobepsychologically,physicallytaxingorwhowereunsureoftheabilities
tocopewithsuchasituationweremorelikelytoexperiencedistress.Fatherswhoalso
madenegativeappraisals,wereunemployedorwhosechildrenwereadmittedtothe
hospitalonaregularbasiswerealsomorepronetopsychologicaldistress(Sloper,
2000).Finally,bothmothersandfatherswerelikeliertopreventpsychologicaldistressif
theywerepartofacohesivefamily(Sloper,2000).
Oneprotectivefactorthatisconsistentlyfoundtobeinfluentialinindividuals’
adaptationinthefaceofadversityissocialsupport.Socialsupportisaresource
comprisingoftheabilityofanindividual’ssocialnetworktoaidtheindividualincoping
withlifestressors(Grootenhuis&Last,1997).Alackofsocialsupporthasbeenfoundto
beassociatedwithpooreradjustmentinparentsofchildrenwithcancer,specificallyin
thedomainsofpsychologicalhealthandabilitytocope(Dockerty,Williams,McGee,&
Skegg,2000).Moreover,asparentsexperienceincreasingpsychologicaldistress,theless
likelytheyaretobesatisfiedwiththeirsocialsupportresources(Hoekstra-Weebers,
Jaspers,Kamps,&Klip,1999).
Parentalcopingstyles.Inlinewiththesefindings,theuseofthesocialsupport
seekingcopingstyleisalsoassociatedwithlesspsychosocialdistressinparentsof
childrenwithcancer(1999).Copingstylesplayanimportantroleinthewaysparents
manage,andasaresult,experiencestressors.Withinthecopingliterature,thereexist
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
17
twomaincopingstylesthathaveproventobeeffectiveincopingwithchronicillness:
Activeandavoidantcoping.Activecopingconsistsofmanagingstressorsinanengaged,
problem-focusedmannerwhileavoidancecopingfocusesprimarilyonemotionalcoping
anddistractionfromthestressor(Kliewer&Lewis,1995).Theseempiricallyvalidand
reliableconstructs(Suls&Fletcher,1985)havebothbeenshowntobeeffectivein
contextsofchronicillnessdespitetheirseeminglycontradictoryprocesses.Ithasbeen
arguedthatavoidantcopingstrategiesareparticularlyeffectiveincontextswhere
individualshavelittlecontrol,suchasinthecaseofchronicillness.Inastudyby
Dahlquist,Czyzewski,Copeland,Jones,Taub,andVaughan(1993),parentsofnewly
diagnosedchildrenwithcancerwerefoundtodemonstratemoreavoidantcopingstyles
thanactivestyles.Theauthorspostulatethatparentsutilizedavoidantcoping
behavioursbecausethesituationwasoutoftheircontrol(Dahlquistetal.,1993).
Moreover,theuseavoidantcopingbehavioursaidedtheparentstoignoreoverwhelming
stressorsandfocusonsurvivingtheshort-termmedicalobstacles.Ithasalsobeen
proposedthatavoidantcopingmechanismsonlybecomemaladaptiveiftheybeginto
interferewithhealthbehavioursoriftheysupportthepresenceofintrusivethoughts
(Shontz,1975;Suls&Fletcher,1985).Conversely,extensiveresearchalsosupportsthe
effectivenessofactivecopingmechanismsinparentsofchildrenwithcancer.Prior
researchindicatesthatparentswhoengagedinactivecopingbehaviourswerelesslikely
todevelopdepressionandanxiety(Norberg,Lindblad,&Boman,2005).Anactivecoping
stylecanalsobeconsideredcrucialinsecuringthemedicalcarenecessaryforachild
withararedisease(Dellve,Samuelsson,Tallborn,Fasth,&Hallberg,2006).
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
18
Samson,Siam,andLavigne(2007)havedevelopedanintegrativemodel
demonstratingthewaysinwhichindividualscopewithillness-relatedstress.Themodel
isacombinationofSamson(2006),CohenandLazarus(1979),MoosandTsu(1977),
Corr(1992)andSamsonandClark(2007)’smodels,asallarepremisedontheuseof
adaptivetasksasameansofcopingwithillness(Samson,Siam,&Lavigne,2007).
Samson,SiamandLavigne’s(2007)IntegratedModelbeginswithacknowledgingthat
everyindividual’scontextisuniqueandshouldthereforebetakenintoconsideration.
Samson,SiamandLavigne(2007)organizecontextualinformationinto3groups:
Personalhistoryandcharacteristics,illness-relatedfactorsandfeaturesofthephysical
andsocio-culturalenvironment.Thesefactorswillinfluencetheindividual’sprimary
cognitiveappraisalofthediagnosis,aconceptinitiallydevelopedbyCohenandLazarus
(1979).Thosediagnosedwillappraisetheirdiagnosistobeeitherbenignorwarranting
stress.Individualswillthenassesstheirresourcestocopewiththediagnosis(secondary
appraisal).Samson,SiamandLavigne(2007)thentheorizethatcertainadaptivetasks
willbeeffectiveincopingwiththediagnosis.Theresearchers,buildingonCorr(1992)’s
adaptivetaskmodel,prescribeadaptivetasksinthefollowingexperientialdomains:
Physical,psychological,social,spiritual,andvocational.Adaptivetasksincludecreating
personalmeaningoftheillnessexperienceandcultivatingsocialsupportgroups
(Samson,SiamandLavigne,2007).Participatingorrefrainingfromengaginginthese
adaptivetaskswillplayalargeroleinthecopingbehaviourutilizedbytheindividual.
Variousstylesandbehaviourshavebeenidentified,withvaryingdegreesofeffectiveness
inillnesscontexts.Copingskillsdifferintheirfocuses,withsomebasedonmeeting
emotionalneedswhileothersfocusonactivelymanagingcontextualcues(Samson,Siam,
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
19
&Lavigne,2007).Thecopingskillsthatareusedwillultimatelymitigatetheillness
experience,creatinga“newstateofpsycho-socialequilibrium”(p.24)orpsychosocial
maladaptation(Samson,Siam,Lavigne,2007).
Experienceofhope.ResearchbySamson,Tomiak,Dimillo,Lavigne,Miles,
Choquette,Chakraborty,andJacob(2009)demonstratesthatthe‘fabricofhope’of
parentsofchildrenwithDuchennemusculardystrophyisstronglyinfluencedbyparents’
cognitiveappraisalsofthesituation,andasaresult,evolves.Theresearchersfoundthat
atthetimeofinitialdiagnosis,parentsappraisedthesituationtobeacrisisandfocused
theirresourcestowardhopingforadvancementsinresearchandtreatments.Asthe
illnessprogressed,theparentsbecomemoreawareoftheirresourcesandwerebetter
abletoredirecttheirfocusfromhopingforacuretoactivelycopingwiththeirchild’s
needs(Samson,etal.,2009).Astheparentsbegintoadapttotheirnewreality,they
begantoallowthechild’sidentitytoflourishratherthanfocussolelyonthedisease.As
medicalcomplicationsbegantoariseanddeathbecomesimminent,parents’hopetook
onaspiritualcomponent(Samsonetal.,2009).
Familydynamics.Parents’psychologicaladjustmenttotheirchildren’sillnessis
predictiveoftheirmaritalsatisfaction(Dahlquist,Czyzewski,&Jones,1996).Although
parentsofchildrenwithcancerdonotexperienceahigherdivorcerate,theydoreportto
belesssatisfiedintheirmarriages(Kazak,1989).Discrepanciesinpartners’levelof
distresshavealsobeenshowntopredictmaritalmaladjustment(Dahlquist,Czyzewski,
Copeland,Jones,Taub,&Vaughan,1993).Dalquistandcolleagues(1996)hypothesize
thatmaritalpartnersrepresentprimaryresourcesofsocialsupportandthereforeplay
animportantroleintheparentalexperienceofraisingachildwithcancer.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
20
Tomiakandcolleagues’(2007)researchontheexperiencesofparentsofchildren
withDuchenneMuscularDystrophydemonstratesthatparentsofchildrenwiththe
conditionmustmakesignificantpsychosocialadjustmentstotheiruniquerealities.The
authorsdiscoveredthatmothersandfathersdifferintheirexperiencesoftheirchild’s
illness,withmothersoftentimesservingasthechild’sprimarycaregiverandfathers
actingassupportingparent(Tomiaketal.,2007).Thisdifferenceinrolescanleadto
discrepanciesinthewaystheparentingofachronicallyillchildisexperienced,suchasin
thewaysparentscopewiththeirchild’sdiagnosisandeverydayobstacles.Mothers
generallyseeksocialsupportasameansofcopingwiththeirchild’sillnesswhilefathers
viewthesituationasaseriesofobstaclestobeovercome(Tomiaketal.,2007).Such
discrepanciesinrolesandexperiencescanaffectthementalhealthofeachpartner
differently,thefamilydynamicandthecareofthechild.
ExperiencesofPolyposisSyndromes
Althoughconsiderableresearchhasbeenconductedontheclinicalandbiological
aspectsofPeutz-JeghersSyndromeandJuvenilePolyposisSyndrome,virtuallyno
researchhasbeenconductedonthepsychosocialaspectsofparentingachildlivingwith
theconditions.Thisdearthofresearchintheliteraturerepresentsanimportantvoidin
ourunderstandingofpolyposissyndromes.AssymptomsofbothPeutz-Jeghers
SyndromeandJuvenilePolyposisSyndromegenerallypresentthemselvesduringyouth,
parentsplayapivotalroleinadvocatingfortheirchildren’smedicalneedsand
supportingtheirchildrenthroughtheexperience(Woo,etal,2009).Aspreviously
discussed,parentsofchildrenwithchronicillnessarevulnerabletopsychological
distress(Kazaketal.,2005;Magni,Carli,DeLeo,Tshilolo,&Zanesco,1983).Peutz-
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
21
JeghersSyndromeandJuvenilePolyposisSyndromearerareandheritableconditions
thataddalayerofcomplexitytoparents’experiences,warrantingfurtherexploration,as
itcannotbeassumedthatthecancerliteratureadequatelyrepresentsthepolyposis
experience.Whilenoresearchhasbeenconductedontheexperiencesofparentsof
childrenwithpolyposissyndromes,reviewoftheexperiencesofindividualswiththese
conditionsmaygiveusinsightintotheissueswithwhichparentsmustcontend.
InastudyconductedbyWooandcolleagues(2009),individualswithPeutz-
JeghersSyndromewerefoundtoexperiencesignificantdistressregardingthepossibility
ofdevelopingcancer.VanLierandcolleagues(2010)alsoreportedthatindividuals’with
Peutz-JeghersSyndromefeltlimitedbyemotionaldifficulties,sufferedpoorermental
healthandratedtheirhealthtobepoorerthanthatofcontrols.Theresearchersfound
participantswhobelievedPeutz-JeghersSyndrometohaveasignificantnegativeimpact
oftheirlivesandwhofeltemotionallyimpactedbytheconditionweremorelikelyto
experienceadiminishedqualityoflife(VanLieretal.,2010).Participantsinthestudy
werealsofoundtosufferfrommilddepression.
Peutz-JeghersSyndromeplayedasignificantroleinparticipants’importantlife
decisions;theconditionwascitedasbeingthereasonmanyparticipantswerehesitantto
havebiologicalchildren-forfearoftransmittingthecondition(Wooetal.,2009).Van
Lierandcolleagues(2012)reportfindingssimilartoWooandcolleagues(2009)’s,ina
studywhereamajorityofparticipantschosenottohavebiologicalchildrenforfearof
transmittingthedisease.Theseparticipantsalsohadahigherincidenceofcancerand
reportedgreaterfearofthedevelopmentofcancerthanparticipantswhodidnotrefrain
fromhavingbiologicalchildren(VanLieretal.,2012).
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
22
Consistentwiththerarediseasesliterature,theresearchersalsodeterminedthat
theparticipantsfelta‘SocietalBurden’,indicatingdissatisfactionwiththemedical
community’sknowledgeandcareofPeutz-JeghersSyndrome(Wooetal.,2009).Sugars
(2011)recountsherfirsthandexperiencewithPeutz-JeghersSyndromeandhercreation
ofaPeutz-JeghersandJuvenilePolyposisSyndromeinternetsupportgroupforthose
affectedbytheconditions,bothdiagnosedindividualsandtheirfriendsandfamilies,asa
meanstosupplementthegapsintheknowledgebythemedicalcommunityandcreating
anorganizedsupportsystem.Partoftheimpetusforcreatingthewebforumwasthe
author’sownexperienceofisolationandfear(Sugars,2011).Furthermore,Sugars
(2011)writesthatamongsttheresearchonPeutz-JeghersSyndromeandJuvenile
Polyposis,thereisalackofresearchdepictingtheexperiencesofthosediagnosedwith
theseconditions.
Coping,animportantmitigatingfactorintheillnessexperiencehasbeenshown
tobesocializedfromparenttochildinfamilieswhereparentsandchildrensufferfrom
thesameillness.InastudybyMiller,Bauman,Friedman,andDeCosse,(1986),
individualswithFamilialAdenomatousPolyposis(FAP)whoseparentswiththesame
conditiontookanactiveroleinguidingtheirchildrenthroughtheillnessexperience,
werelesslikelytoexperienceanxietyabouttheirfuturehealth.Kliewer,Fearnowand
Miller(1996)havedevelopedamodelinwhichcopingstrategiesaretransmittedfrom
parenttochild,basedonresearchinanon-clinicalpopulation.Theresearchersinclude
parentalcopingsuggestionsandparentalmodelingasmeansoftransmission,with
influencesfromfamilydynamics(Kliewer,Fearnow,&Miller,1996).Intheirstudyusing
questionnaires,theauthorsfoundthatmothershaveagreaterimpactontheirchildren’s
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
23
copingskillsthanfathers(Kliewer,Fearnow,&Miller,1996).Thequalityoftheparent-
childrelationshipwasalsofoundtobeassociatedwithstrongercopingsocialization
effects(Kliewer,Fearnow&Miller,1996).Thetransmissionofcopingmethodsin
hereditarydiseasessuchaspolyposissyndromesposesauniquecomplexityinillness
experiences,asparents’ownexperiencewithillnesswilllikelyinfluencetheirparenting
behaviorsoftheirchildrenwiththesameillness.
Insum,theliteratureavailableonrarediseases,experiencesofparentsof
childrenwithchronicillnessandexperiencesofpolyposiscreatesastrongrationalefor
furtherresearchintotheexperiencesofparentsofchildrenwithpolyposissyndromes.
Whilethelinesofresearchonparents’experiencesofraisingchildrenwithcanceris
currentlypractitioners’mostrelevantresource,itcannotbeassumedthatthesecontexts
aregeneralizabletothepolyposisexperience.Becauseoftheuniquepropertiesof
polyposissyndromes,italsocannotbeassumedthatpriorresearchortheoretical
frameworksrepresenttheexperiencesofindividualswithchildrensufferingwith
polyposissyndromes.Cancerresearchhasrevealedcommonthemeswithinthecancer
experience,suchasexperiencingstigma(DiMilloetal.,2013),andfearofdying(Murray,
Grant,Grant,&Kendall,2003)thatcannotnecessarilybeattributedtopolyposis
syndromeswithoutfurtherresearch.Assuch,thisexploratory,qualitativestudy
presentsanopportunityforresearcherstostrengthenthepolyposisliteratureandmore
effectivelyengageclinicalpractitioners.
ObjectivesandResearchQuestions
Themainobjectiveofthisstudyistoexploretheexperiencesofparentsof
childrenandteenagersundertheageof20(hereinafterreferredtoas‘children’)with
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
24
polyposissyndromesusingahermeneuticphenomenologicalapproach.Thefollowing
researchquestionwilldirectthisexploratorystudy:Whatarethelivedexperiencesof
parentsofchildrenwithpolyposissyndromes?
ContributionstoKnowledgeandPractice
Theresearchliteraturedemonstratesaclearvoidinknowledgeaboutthe
experientialimpactofparentingachildwithapolyposissyndrome.Theresultsfromthis
studyandthemeansbywhichitiscollectedprovidetheresearchliteraturewith
invaluableinformation.Furthermore,thefindingsgeneratedbythisstudyaidphysicians
andpsychosocialpractitionersworkingwithparentsofchildrenwithpolyposisto
understandthecomplexitiesoftheirlivedexperiencesandultimatelyofferbettercareto
bothpediatricpatientsandtheirparents.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
25
CHAPTER3:Methodology
Theliteraturereviewprovidescontextforthisstudy.InthischapterIdiscussthe
philosophicalfoundationsguidingthisstudyaswellasthehermeneutical
phenomenologicalapproachIused.Furthermore,Idelineatemypositionasaresearcher.
IalsodiscussthestrategiesIusedtodevelopmydatacollectioninstrumentsaswellas
thewaysIrecruitedparticipants,collectedthedata,andanalysedit.Lastly,Ihighlight
thestepsItooktoestablishmethodologicalrigour.
Thisstudywasconductedusinganinductive,qualitativehermeneutic
phenomenologicalapproach.ThisapproachwasinformedbyLindsethandNorberg
(2004)’sPhenomenologicalHermeneuticalMethodforResearchingLivedExperience.
Thematicanalysisoftheparticipants’transcribedinterviewswasundertakentodevelop
torevealthecommonessencesoftheparticipants’sharedexperiences.Asidentifying
essencesisthe“methodologicalgoal”ofphenomenologicalresearch,theessences
revealedinthisresearchserveasthestudy’sfindings(Dahlberg,2009,p.11).
PhilosophicalFoundations:HermeneuticalPhenomenology
Phenomenologicalresearchseekstorevealthe“essenceorstructureofan
experience”(Merriam,2002,p.7).Oneofthecentraltenetsofhermeneutic
phenomenologyisthat“complexmeaningsarebuiltoutofsimpleunitsofdirect
experience”(Merriam,2002,p.7).Hermeneuticphenomenology,developedbyMartin
Heidegger,buildsontheworkofEdmundHusserl,thefatheroftranscendental
phenomenology.Husserl(1931)positedthatphenomenologistsshouldengagein
bracketingwhenexploringaphenomenon,wherebytheresearchersattempttosuspend
theirownbiasedviewsoftheworldinordertoobserveandanalysetheexperiencein
questioninitstruestform.Heidegger,astudentofHusserl,deviatedfromthe
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
26
transcendentalphenomenologicalviewpointstodevelophermeneuticphenomenology,
whereinphenomenologistsarebelievedtobeunabletosuspendtheirbiasesand
thereforeareencouragedtofocustheirattentiontotheawarenessoftheirinfluenceon
theexploration(Connelly,2010).Rather,Heidegger(1962)arguedthataresearcher’s
priorknowledgecanbebeneficialtotheresearchprocessandcanactuallyenrichthe
researchprocess.Heidegger(1962)alsohighlightedthenotionofco-constitutionality,
whereinbothparticipantsandresearchersareviewedaspartnersindevelopingand
interpretingmeaningfromparticipants’narratives(Koch,1995).However,hermeneutic
phenomenologistsmustensurethatthederivedmeaningsarereflectiveofparticipants’
experiences,well-reasoned,andreflecttheimplicationsofthemeaningsinvarious
practicalfields(Annells,1996).
In exploring the various manifestations of the phenomenological approach, I
reflectedonmyownepistemologicalandontologicalviewpoints,aswellasmyownrole
as researcher.Upon reflection, I realized thatmybiases, views, andexperiencesmight
influencemyinterpretationofthedata.Assuch,Icontinuedtoengageinself-reflection
anddocument theways inwhich I felt I influenced theprogressionof this study.This
reflectionservedtoenrichtheresearchprocess.
RationalefortheuseoftheHermeneuticalPhenomenologicalApproach
Asmentioned,thisstudywasconductedusingahermeneuticalphenomenological
approach.Thisapproachallowstheresearchertoplayanactiveroleincollecting,
organizingandinterpretingdatainawaythatisreflectiveofthelivedexperiencesofthe
parentsofchildrenwithpolyposissyndromes.Ihavechosenthephenomenological
approachtoguidethedevelopmentandactualizationofthisresearchbecauseofthe
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
27
focusoftheapproachontheinterpretationofsharedlivedexperiences.Iappreciatethe
recognitionthatthisapproachhasforcontextinsubjectiveexperiences.AsIam
choosingtodescribeandinterpretthelivedexperiencesoftheparentsofchildrenwith
polyposisdiagnoses,Ibelievethatthisapproachisbestsuited.
Alimitedamountofqualitativeresearchhasbeenconductedinthedomainof
parentalcopingofchildrenwithchronicillness,withthemajorityofstudiesemploying
quantitativeanalysisusingstructuredquestionnaires.Thesoleuseofempirical
methodologiesasameanstorepresentingcomplexexperiencesriskslosingimportant
experientialdetailsindatarepresentedbynumbers.Theuseofqualitativedatain
understandingtheexperiencesofparentsofchildrenwithpolyposissyndromesis
integral.Polyposissyndromesdemonstratevariousuniquepropertiesdissimilartomany
illnessesstudiedintheliterature.Peutz-JeghersSyndromeandJuvenilePolyposis
Syndromearerareconditionsthatstronglypredisposethosediagnosedtocancer.
Moreover,theseconditionsarehereditaryandchronic.Itisnotyetknownhowthe
combinationofthesefactorsaffectstheexperiencesofparentsinthecontextof
polyposissyndromes.Manyinterrelatedfactorsplayaroleintheexperiencesofparents
ofchildrenwithpolyposissyndromesandthehermeneuticalphenomenological
approachallowsforthemostintegrativeandmeaningfulcollectionandanalysisofthese
factors.Whileresearchontheexperiencesofparentsofchildrenwithcanceriscurrently
ourgreatestsourceofinformationintotheexperiencesofparentsofchildrenwith
polyposis,itcannotbeassumedthatthesephenomenacanbegeneralizedtopolyposis
experiences.Becauseoftheuniquepropertiesofpolyposissyndromes,italsocannotbe
assumedthatpriorresearchrepresentstheexperiencesofindividualswithchildren
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
28
sufferingwithpolyposissyndromes.Assuch,hermeneuticalphenomenologyservesasa
usefultoolinbeginningtheexplorationoftheseexperiences.
Hermeneuticalphenomenologypositionsparticipantsastheexpertsoftheir
experiences,animportantfeatureofqualitativeresearch(Marques&McCall,2005).
Participantswillbeabletofocusonthemesmostsalienttothem,therebyallowingthem
tocreatetheirownnarratives.AsIamseekingtodescribeandunderstandthemeaning
behindtheparents’experiencesoftheirchildren’sillnesses,hermeneutical
phenomenologyservesasanappropriateapproachasitwillallowmetodelveand
explorethelivedexperiencesoftheseindividuals(Creswell,2013).
ResearcherPosition
Priortothestartofmygraduatestudies,Iworkedwithahealthpsychology
researchgroupthatstudiedtheexperiencesandhealthbehavioursofindividualswith
cutaneousmelanoma.Suchexperienceignitedmypassionforhealthpsychologyand
interestinthewaysinwhichindividualscopewithadversity,specificallyhealth
challenges.Asaninterncounsellorduringmygraduatestudies,Ilearnedthedepthsto
whichcontext,environmentandpsychologicalfactorsplayincreatingexperienceand
mitigatingcopingstrategies.Furthermore,mytrainingincounsellinghasallowedmeto
beabletoeffectivelyconveyempathyandencourageclientstospeakopenlyabouttheir
experiences;askillIfeelwaseffectivelyutilizedinthedatacollectionprocess.I,myself,
donotsufferfromapolyposiscondition,noramIaparenttoachildwithsucha
diagnosis.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
29
Participants
Eligibilitycriteria.Inordertobeeligibletoparticipateinthisstudy,participants
hadtobetheparent,caretaker,guardian,orindividualservingwithinaparentalroleofa
pediatricpatientundertheageof20yearsdiagnosedwitheitherPeutz-Jeghers
SyndromeorJuvenilePolyposisSyndromeatthetimeofthestudy.Bothclinicaland
moleculardiagnosesoftheseconditionswereaccepted.Participantsmustnothavehada
psychiatricormedicalillnessthatwouldpreventthemfromparticipatinginthe
intervieworgivinginformedconsent.Participantsneededtobeabletocommunicatein
eitherFrenchorEnglish,asinterviewswereonlyconductedineitheroftheselanguages.
Participantsmeetingthesecriteriawererecruitedforthestudy.
RecruitmentProcess.PhysiciansatapediatrichealthsciencescentreinOntario,
identifiedparticipantsfromtheirpatientlistsfittingtheeligibilitycriteria.Oncethelist
ofeligibleindividualswascompleted,participantswerecontactedandinformedabout
thestudy.Bothparentsofeachpediatricpatientwereinvitedtoparticipate,allowingfor
bothamotherandfatherofthesharedchildtoparticipate.Participantswererecruited
usingacriterionsamplingmethod,whereineligibilityforthestudywasdependenton
havinglivedtheaforementionedphenomenon.Asdiscussed,smallpopulationsizesare
aninherentchallengeofstudiesstudyingrareillnesses(Ettore,2006).Oncetheeligible
patientsandtheirparentswereidentified,thephysiciansaddressedandmailed
envelopescontainingtheStudyDescriptionandPermissiontoContactParticipantsforms
(AppendicesBandC,respectively),aswellasinstitution-addressedstampedenvelopes
sothattherecipientscouldeasilymailbacktheirresponses.Thephysiciansnotified
myself,theprimaryresearcher,onceshereceivedthereturnedenvelopes,atwhichpoint
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
30
Icontactedparentsconsentingtobecontactedbytheirpreferredmeans,eitherby
telephoneorbyemail.Inthefollow-upcontact,Iinformedparentsabouttheobjectives
ofthestudyandwhattheirparticipationwouldentail.Oncetheirquestionswere
answered,parentswereinvitedtoparticipateinthestudy.Ithenscheduledadateand
timetomeetatthepediatrichealthcentretoconducttheinterview,atwhichpointthe
ParticipantInformationandConsentForms(AppendixD)werecompleted.
Parentswhodidnotrespondtotherecruitmentletterswithinthreeweekswere
sentareminderrecruitmentpackagecontainingthesamedocuments.Intotal,thirteen
individualparents(ofelevendiagnosedchildren)werecontactedtoparticipateinthis
study,withsevenparentsindicatingtheirinterestandconsenttoparticipateinthe
study.Allparentswhowerecontactedregardingthestudyweresentboththeinitial
recruitmentformsaswellasthefinal,identicalreminderrecruitmentforms.Theparents
whowerecontactedbutwhoultimatelydidnotparticipateinthestudyindicatedtheir
disinterestinparticipatinginthestudybynotrespondingtoeitherofthetwocontact
attemptsforrecruitment.
InstrumentDevelopment
DevelopmentoftheInterviewProtocol.Theinterviewprotocolwascreatedby
consultingrelevantresearchandmedicalprofessionalsworkinginthegastroenterology
andgeneticsfields.Althoughverylittleresearchhasyettobeconductedonthe
experiencesofparentsofchildrenwithpolyposissyndromes,findingsofrelevant
researchindicatethatparentsofchildrenwithchronicillnessessufferanincreasedrisk
ofmentalhealthandemotionalproblems(Dalquist,Czyzewski,&Jones,1996;Kazak,
Boeving,Alderfer,Hwang,&Reilly,2005;Sawyer,Antoniou,Toogood,Rice,&Baghurst,
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
31
1993).Asameansofmanagingthestressconferredbyraisingachildwithchronic
illness,parentsgenerallyturntotheirpartners,withanincreasedlikelihoodof
influencingfamilydynamics(Budych,Helms,&Schultz,2012;Dellve,Samuelsson,
Tallborn,Fasth,&Hallberg,2006).Thenewlyemergingliteratureonrarediseasesalso
demonstratesindividualswithrarediseasesundergosignificantlydifferentexperiences
aspatientsthanindividualswithmorecommonconditions,aspatientsbecomeexperts
ontheirdiagnosesandtheirownbiggestadvocates(Aymé,Kole,&Groft,2008;Budych,
Helms,&Schultz,2012).Assuch,questionswerecompiledspecificallytoexplorethese
domains(i.e.Psychologicalimpact,socialimpact,medicalcareseekingexperience,
adaptationtoillness,viewofchild’shealth,familyplanningdecisions)
Althoughthereexistsresearchonthevariousinfluentialfactorsofbeingaparent
ofchildwithchronicillness,itcannotbeassumedthatsuchfindingsaregeneralizableto
parentsofpolyposissyndromes.Theinterviewprotocolensuredparticipantswiththe
flexibilitytofocusonparticularlysalientissuesintheirexperienceswhileprobing
evidence-basedtopics.Interviewquestionswereorganizedandorderedtofacilitatethe
developmentoftrustbetweentheresearcherandparticipant.Open-endedquestions,
humanisticinnature,werealsousedinordertopromotetheparticipants’introspection
andexplorationoftheirexperiences.
DevelopmentofDemographicQuestionnaire.Participantswereaskedto
completeademographicquestionnaireinordertoenabletotheresearchertomore
appropriatelycontextualizeandunderstandthecollecteddata.Variablesassessedwithin
thedemographicquestionnairewereincludedbasedontheirdemonstratedrelevancein
priorresearch.Participantswereaskedtolisttheresourceswithintheirsupport
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
32
networksinordertobetterunderstandtheirsocialsupportexperiences.Furthermore,
theparentswereaskedtodescribetheirmaritalstatusesandtolistthenumberof
childrentheyparentinordertogainfurtherinsightintotheirfamilysituation.Lastly,
participantswereaskedtospecifydetailsoftheirownandtheirchildren’smedical
historiesinordertobuildarichdescriptionofthesamplegroup.
Demographics.Asmentioned,atotalofsevenparticipants(representingsix
patients,astwooftheparticipantshadformerlybeenmarried)wererecruitedtotake
partintheinterviewprocess.Allparticipantsatthetimeoftheinterviewweremarried,
exceptforoneparticipantwhoidentifiedhimselfasasingle-parentwidower.Combined,
theparticipantshadanaverageoftwochildren,witharangeofonetofourchildren.All
oftheparticipantshadeitheroneortwochildrendiagnosedwithJuvenilePolyposis
Syndrome,exceptforoneparticipantwhosechildwasdiagnosedwithPeutz-Jeghers
Syndrome.NoneoftheparticipantssufferedfromJuvenilePolyposisSyndromeorPeutz-
JeghersSyndromethemselves.OnlyoneparticipantindicatedthathisJuvenilePolyposis
diagnosedchildrenhadbeenidentifiedashavingafamilyhistoryofthecondition;his
wifehadpassedawayfromcomplicationsrelatingtothecondition.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
33
Table1
Participants’Demographics
Item n(%) Sex Male 2(29)Female 5(71) Primarylanguageofcommunication English 4(57)French 3(43) MaritalStatus Married 6(86)Single 1(14) Highestlevelofeducationachieved HighSchool 1(14)CEGEP/TechnicalCollege 2(29)University(Undergraduatedegree) 2(29)University(Graduatedegree) 2(29) Participantdiagnosisofpolyposiscondition 0(0) Numberofchildren 1 1(14) 2 3(43) 3 1(14) 4 2(29) Numberofchildrenwithpolyposisdiagnosis 1 5(71)2 2(29) Polyposisdiagnosisofchildren JuvenilePolyposisSyndrome 6(86)Peutz-JeghersSyndrome 1(14) Useofgenetictesting 7(100) Individualspartofsupportsystem Partner/spouse 5(71)Family 5(71)Friends 5(71)Physicians 5(71)Co-workers 0(0)Psychologicalservices 1(14)Internetforums 1(14)
DataCollection
Participantsmettheprimaryresearcheratahealthsciencesinstitution,where
theywerebroughttoaprivateroomlocatedintheGastroenterologyunit.Thislocation
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
34
ensuredthatparticipantswereaffordedprivacyandconfidentialityastheydiscussed
theirpersonalexperiences.Thesitealsoservedasafamiliarsitetoparticipants,
minimizingunduestressconcerningattendingtheinterview.
Priortocommencingeachinterview,theresearcherreviewedtheParticipant
InformationandConfidentialityForm(AppendixD)andensuredtheparticipant
understoodtheinterviewprocess.Shethenwitnessedtheparticipantsigntwocopiesof
theParticipantInformationandConfidentialityForm(AppendixD),oneofwhichwas
keptbytheresearcher,andonekeptbytheparticipantfortheirfiles.Participantswere
reimbursed$20.00fortheirparkingfeesbytheresearcher.Participantswerethenasked
tocompletetheDemographicInformationForm(AppendixA).Theinformationcollected
fromthisdocumentwasusedtocreatethecontextwithwhichparticipants’datawas
interpreted.
Datawascollectedthroughtheuseofthesemi-structuredInterviewProtocol
(AppendixE).Thesemi-structuredinterviewprocesswaschosentoaffordthe
interviewertheflexibilitytoprobeparticularlysalientthemeswithparticipants.The
effectivenessofsemi-structuredinterviewswasalsoenhancedasitwasusedwith
concurrentdataanalysisandcollection,whichallowsforinitialinterviewstoinformthe
organizationofsubsequentinterviews.Interviewsweredigitallyrecordedand
transcribedverbatimasameansofensuringaccuraterecordsandanalyses.Allfiles
werekeptontheprimaryresearcher’spassword-protectedcomputer.Participants’
identitieswereprotectedbypseudonym,withidentity-revealinginformationomitted
fromfinaltranscripts.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
35
Interviewrecordingswereimmediatelytranscribedaftereachinterviewsession
inorderfortheresearchertobestrecalltheinterviewsessionasawhole.Aftereach
transcribingprocess,theresearcherhighlightedandtooknoteofpossiblethemesthat
shouldbeaddedtosubsequentinterviewprotocolsintheformofquestionsorprobes.
DataAnalysis
Aspreviouslydiscussed,thehermeneuticalphenomenologicalapproachdoesnot
prescribeaspecificmethodology(Colaizzi,1978;Hein&Austin,2001).Thedata
collectedforthisstudywasanalyzedusingLindsethandNorberg(2004)’soutlined
approachtohermeneuticalphenomenologicalanalysis.Ichosethistypeofanalysisasit
wasspecificallydesignedtoexplorelivedexperienceswithinhealthcarecontexts
(Lindseth&Norberg,2004).Theauthors’structureforanalysisaimsto“elucidate
essentialmeaningasitislivedinhumanexperience”(Lindseth&Norberg,2004,p.146).
Suchelucidationofmeaningisrevealedthroughstory-tellingandexpressionofone’s
experiences(Lindseth&Norberg,2004).LindsethandNorberg(2004)alsoarguethat
interpretationofthelivedexperiencesshouldbedoneusingtextualmaterial
(transcribedcopiesofparticipants’recordedinterviews)throughtheuseofthematic
analysis.
FollowingLindsethandNorberg(2004)’sguidelines,Ibeganthedataanalysis
processbytakingmeticulousnotesduringtheinterviewprocess.Withinhermeneutical
phenomenology,itisunderstoodthattoacertainextentinterviewershelptoshapethe
finalnarrativeproduct(Lindseth&Norberg,2004).OnceIbeganthedatacollection
process,recordedinterviewsweretranscribedverbatimasquicklyaspossibleinorder
tomosteffectivelycaptureeachparticipant’snarrativethroughtext.Datawascollected
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
36
andanalysedconcurrently,withdatafromearlierinterviewsinformingtheformation
andorganizationofsubsequentinterviewquestions.Oncealltheinterviewswere
transcribed,IengagedinwhatLindsethandNorberg(2004)callanaïvereading,
whereinIreadthetranscriptsmultipletimesinordertounderstandthemeaningofthe
narrativesasawhole.
OncethenaïvereadingwascompletedandIfeltthatIhadgainedinsightintothe
meaningoftheparticipants’experiences(i.e.,theirlifeworlds),Ibeganthestructural
analysisofthedataanalysisprocess.Whilephenomenologicalstructureanalysiscanbe
achievedthroughvariousmethods,Ichosetoengageinthematicstructuralanalysis.
LindsethandNorberg(2004)describetheidentificationandformulationofthemes
(unitsofmeaning)as“methodicalinstancesofinterpretation”(p.149).Wholesentences,
fragmentsofsentences,andparagraphsthatrevealedcommonsmeaningsweregrouped
togetherandrevised,refined,untiltheywerereflectiveandconsistentwiththe
understandingbuiltfromthenaïvereading.Thethemesweresituatedwithinthe
phenomenon–thatisthelivedexperiencesoftheparticipants.Thethemeswerethen
onceagainrefinedusingexperientialandcolloquiallanguage.Mythesissupervisor
servedasanauditorofthedataanalysisprocessandengagedinthesamedataanalysis
processasme.Whendifferenceswerefoundbetweenourfindings,wethenreconciled
ourdiscrepanciesthroughdiscussion.
MethodologicalRigour
Thisstudymaintainedandexaminedmethodologicalrigourusingtheconcepts
andstandardsoutlinedbyLincolnandGubatoachievetrustworthiness(1985):
credibility,transferability,dependabilityandconfirmability.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
37
LincolnandGuba(1985)’sconceptoftransferabilityisdefinedastheabilityto
applytheresultsofonecontexttoanothercontext.Inordertodoso,researcherswould
needthedetailedinformationoftheoriginalcontextinwhichthestudywasdeveloped
(Lincoln&Guba,1985).Asthesamplegroupcomprisesofindividualsexperiencinga
veryuniqueandrarephenomenon,transferabilityofresultsmaybedifficulttoacquire.
However,backgroundcontextsoftheparticipantswereassessedinthedemographic
questionnaire,thedataofwhichhasbeenincludedinthismanuscript.Furthermore,the
manyinterviewquestionswereformulatedinawaytoencourageparticipantstodiscuss
histories,contexts.Theliteraturereviewincludedinthismanuscriptwasalsowritten
withthegoalofbeingabletoeasilysituatethisstudywithinthecurrentavailable
research.
Inordertoensurethecredibilityofthedataobtained,astandardthataimsto
ensurethecongruenceofresultswithreality,establishedqualitativeresearchmethods
werefollowed,medicalspecialistsworkingwithpolyposisfamiliesaswellasrelevant
literatureswereconsultedinorderformetofamiliarizemyselfwithsomeoftherealities
andexperiencedfacedbypatientsandtheirfamiliespriortodatacollection.The
interviewprotocolwasalsoformulatedtoencourageparticipantstospeakhonestly,
withtheinterviewsummarizedandrecountedbacktotheparticipanttoensurethatthe
interviewerunderstoodandinterpretedtheirresponsescorrectly.Myabilitytofollow
thedirectionofemergingthemespresentedbytheparticipantenrichedtheinductive
processanddelvedintothecomponentsoftheexperiencemostpertinenttothe
participants.Furthermore,agastroenterologistwasaskedtoreviewthethemesrevealed
inthisstudytoassessthecredibilityoftheinterpretations.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
38
Thedependabilityoftheresultsisstrong,asthestepstakentodevelopthe
researchdesignandprocedurehavebeenoutlinedingreatdetail.Lastly,inorderto
increasethelevelofconfirmabilityofthisstudy,theuseofasecondresearcherauditing
thecodingprocessservedtolimitthepotentialforbias.Theprimaryresearcherhasalso
providedastatementindicatingherpositioningasaresearcherandpossiblebiasesthat
mayhaveaffectedtheprogressionoftheresearchprocess.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
39
Chapter4:Findings
Thischapterdiscussestherevealedessencesofparticipants’sharedexperiences.
Asessenceswererevealedusingthematicanalysis,theterms‘essences’and‘themes’will
beusedinterchangeably.Thedataanalysisprocessrevealedthatwhiletheparticipating
parentsmayhavecomefromvaryingbackgrounds,theirlivedexperiencessharedmany
commonalities.Theanalysesrevealthatthesesharedcommonalities–essences-are
essentialtothephenomenonofparentingachildwithapolyposissyndrome.Inthis
chapter,Iidentifyanddescribetheessencesrevealedinthedataanalysisprocess.
UseofIdentifiers
Inordertoretaintheanonymityandprotecttheprivacyofthestudy’s
participants,participantnameswerereplacedwithnumberedidentifiers,suchas
‘Participant1’andallidentifiableinformationhasbeenremoved.
DescriptionofEssences
Intotal,fourthemesandtwelveunderlyingsub-themeswererevealed.The
followingthemescomprisetheshared,meaningfulexperiencesoftheparticipantsfrom
thetimeoftheirchildren’sdiagnosesonward:(1)Buildinghealingrelationshipswithin
themedicalcommunity(a.Comingtogetherforthewellbeingofthechild;b.Alwayson
thelookout:Doingeverythingpossibletomaintainhealth;c.Feelinggrateful:Itcouldbe
worse);(2)Peekingthroughcoveredeyesatillnessliterature(a.Seekingvalidationin
sharedexperiences;b.Tuningoutanxiety-inducinginformation);(3)Perserveringuntil
needsaremet(a.Teachingthechildrentospeakforthemselves;b.Trustingmaternal
intuition;c.Takingitonedayatatimebecausethefutureisuncertain);(4)Creatinga
new‘normal’aseverythingbecomesunsettled(a.Becomingadifferenttypeofparent;b.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
40
Trustingtheywillspeaksotheycanbeheard;c.Puttingfamiliestothetest:copingwith
theillnessasafamily;d.Feelingasthoughnooneunderstands).Diagramsareincluded
withinthechaptertovisuallyrepresenttherelationsbetweenthethemesandsub-
themes.Figure1,shownbelow,depictsthesub-themescomprisingthe‘Buildinghealing
relationshipswithinthemedicalcommunity’theme.
Figure1.VisualrepresentationoftheBuildinghealingrelationshipswiththemedicalcommunity
themeandsub-themes
Buildinghealingrelationshipswithinthemedicalcommunity
Thefollowingthreesubthemeswerefoundtocontributetotheunderstandingof
thetheme“Buildinghealingrelationshipswithinthemedicalcommunity”:(a)Coming
togetherforthewellbeingofthechild;(b)Alwaysonthelookout:Doingeverything
possibletomaintainthechild’shealth;(c)Feelinggrateful:Itcouldbeworse.Inthis
section,Idiscusshowthephysicians’wayofinteractingandrelatingwiththepatients’
parentsultimatelyshapetheparents’cognitions,emotionsandbehaviours.Astheir
children’sconditionsprogressed,parentsreportedfeelingheardandincludedinthe
Feelinggratitude:Itcouldbeworse
Buildinghealingrelationshipswithinthemedicalcommunity
Comingtogetherforthewellbeingofthechild
Alwaysonthelookout:Doingeverythingpossibletomaintainthechild’shealth
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
41
medicalprocessbytheirchildren’sphysicians,theydescribedfeelinglessanxietyand
uncertaintyabouttheirchildren’shealth,moregratitudeforthehealthoftheirchildren
aswellastheavailablemedicalresources,andweremorecommittedtoensuringtheir
childrenwereappropriatelyfollowedbythemedicalteam.
ComingTogetherfortheWellbeingoftheChild.Whendescribingtheirlived
experiences,parentsdescribedtheirrelationshipwiththeirchild’sphysiciantobe
particularlyinfluentialinaffectingthewayinwhichtheycopedwiththeirchild’s
diagnosis.Whilesomeparticipantsdiscusseddifficultinitialexperienceswiththeir
familyphysicians,theremainderoftheparticipantgroupdiscussedpositiveexperiences
withtheirchildren’sphysicians.Theydescribedrelationshipsinwhichphysicians
engagedtheminthecollaborativecareoftheirchild.Bycollaboratingwithparentsto
ensuretheirpatientsreceivedthemostappropriatemedicalcare,physicianswereable
tobuildfoundationsoftrustwithparents.Forexample,participant5explainedthatin
hermeetingswithherchild’sgastroenterologist,thatshefeltcomfortableengagingthe
doctorinadiscussionaboutherconcernsforherchild.Shestated:
Well,um,Ifeltverysafe.Ifeltverycomforted.Ifeltfreetovoicemystress,my
angst,myanxiety,myconcerns.IfeltlistenedtoandIfeltheard.Ifeltthiswasa
verysafeplacetohaveallthesediscussions.Um,Imeantothepointwherewe,
myex-husbandandI,hadanopenandcandidconversationwith[thedoctor]
aboutourmaritalbreakdownandwhetherthestressofthatmayhaveimpacted
ourchild’shealth.Andshedidnotshyawayfromthatconversation,wedidn’tshy
awayfromthatconversation.Itwasjustaveryopenandhonestenvironmentto
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
42
considereverythingthatmightimpactthischild’shealth.Itwasreallyacollective
effort.
Participant1alsoreflectedhavingasimilarexperience,inthathealsofelt
comfortableaskingquestionstothedoctor,whichhelpedtoreducehisanxiety.He
explained:
Interviewer:Etpourvous,êtresuivitpar[lemédecin],c’estlameilleurefaçonde
diminuerl’anxieté?
Participant1:Oui,c’estplusrassurantquandtusaisqu’ilssontsuivitdeprof.Elle
faittoutetempsàl’estomac,lesintestine,êtresurqu’ilyariennulpart.Ohoui,
c’estpasmal…Non,c’esttranquille.
Interviewer:Etestcequevousvoussentiezconfortabledeposerdesquestions
[aumédecin]?
Participant1:Oui,oui.
Workingwithinacollaborativerelationshipwithphysiciansalsopromoted
honestyandanopportunityfordialogueonthepartofparentswhowereunhappywith
physicians’recommendations,asParticipant7revealedlearningwhileservingasan
advocateforherson.Sheencouragedparentsinsimilarsituationstoensurethe
physiciansaremadeawareoftheirconcernsandtocreatedialoguesevenwhendoctors
arehesitant.Sheexplained:
Anddon’talways,I’mlearningnowyoudon’thavetodowhat[thedoctors]want
youtodo.Youdon’thavetotakeallthisgarbage[medication]ifyoudon’twantto
takeit.Findmeanothersolution.Findmeabettersolution.‘Idon’thavetodo
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
43
that’.‘Well,thehealthsystemsaysthatyouhaveto.’‘Okay,well,findmeanother
solutionbecausethisoneisn’tworkingforme’.
Inthreeofthesevenparent-physicianrelationshipsdescribed,participants
describedexperiencingdifficultchallengesintryingtoconveytotheirfamilyphysicians
theseriousnessoftheirchildren’ssymptoms.Inthesecases,participantswereplacedin
positionswhereadvocatingfortheirchildren’sneedsprovedtobenecessary,asthe
children’sconditionswererevealedtobelife-threatening.Thefindingsdemonstratethe
lackofapatientandfamily-centeredapproachnegativelyimpactedtheexperiencesof
theparentsandtheiraffectedchildren.
Participant2describedherinitialexperienceintryingtolearnmoreabouther
daughter’shealthassuch:
Well,forsometime,sinceshewasinaboutgradesix,soabouttwelve,she
hadn’tbeenfeelingthatgreat.Wehadgonetothefamilydoctoracoupleof
timesandyouknow,sometimestheyjustsortof,youknow,poopooyou.
Youknow,it’slike,“Ohwell,youknow,she’sbecomingateenager,and
they’rejustirritableortheytendtogettiredalot”,becauseshewastireda
lot.And,um,shewaseatingtonnesandtonnesofice.Justlikecupaftercup
aftercupofice.AndIkeptsayingtothedoctor,“Thisisnotnormalfor
someonetoeatthisamountoficeandtobethistiredallthetime”.AndI
wasignored.
Theparticipantfurtherdescribedmeetingwiththedoctormultipleothertimes,
eventuallyconvincingthedoctortoallowherdaughtertoundergotesting.Ultimately,
theparticipant’sdaughterwasfoundtobeseverelyanemicandnecessitatingemergency
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
44
surgeryduetoapolypblockinghercolon.Whenaskedhowshefeltabouttheexperience
inseekingmedicalcareforherchild,sheexplained:
Well,itwasmostlyatthetimeasenseoffrustrationthatnoonewaslisteningto
us…Sheswitchedfamilydoctorsafterthatbecauseshefeltthatour,thefamily
doctorthatIstillhave,haddroppedtheballonher.
Participant3experiencedasimilarsituation,whereinherfamilyphysicianalso
discountedherconcernsaboutherchild’shealth.Shestated:
AndthensoItookhertothedoctor.IrememberIwasreally,reallysick.Ihada
doubleearinfectionandIhadalittlebaby,likea3-monthold.AndIgotintoabig
fightwiththedoctorbecauseIwantedhertested,Iwantedherbloodworktested.
Isaid“Ithinkshe’sanemic”.AndhetoldmeIwouldbelaughedoutof[the
hospital],werehisexactwords.Hesaid,“I’veseenanemicchildrenandthischild
isnotanemic”becauseshewaswired.Shewasrunningaroundandthiswasa
walk-inclinicandshesaid,hesaid,“You’regonnabelaughedoutof[the
hospital]”.SoIsaid,“Canyoujustgive…”andIliterallyfoughtwithhim.Isaid,
“I’msosick,canyouplease?”ThankGodheagreed,becausehealmostwasn’t
goingtogiveittomeandthenhegave…Igotmyhusbandtotakeherin,Ithink
thenextday,hegotabloodwork.Thenthatcliniccalledusandtolduswehadto
bringherimmediatelyto[thehospital]becauseshewassoanemic,shemight
needabloodtransfusion.
Theparticipantthenstatedthatshe“calledthatdoctorand[she]gavehimapiece
of[her]mind.Andhetold[her]itwasthebiggestlessonofhismedicalcareer.”
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
45
Thesameparticipant,whenmeetingwithherotherchild’sphysician,onceagain
hadtoadvocateforherchild’sneedstoanunreceptiveaudience.Sheexplained:
Asathirdroadblock,whenIwenttomyfamilydoctor,Isaid“[Mydaughter’s]got
polyposis”…[Myyoungerson]isanemic,[he]hasgotbloodinhisstool,[he]has
gotallthesamestuff,youknow,canyoupleasereferhimto[the
gastroenterologist]?Andshegavemeahardtime.“Well,youknow,ifitwasn’tfor
[yourdaughter],Iprobablywouldnot.I’dprobablyjustautomatically…”This
shouldbeano-brainer…Heshouldhavebeenherebefore.AndsoIhadtolike
“Please,canyou,youknow,againstyourbetterjudgment,pleasesendhimto
Dr…?”Like,yeah,itreally…Idon’tknow.Idon’tknow.
Inbothexperiences,thephysicians,whenfirstdiscussingthepatients’symptoms,
didnottakethemothers’concernsseriously.InthecaseofParticipant2,itisparticularly
evidentthatthephysiciandidnotviewtheparticipant’sexperiencesofherchildren’s
symptomsasvalid,positioningherselfastheexpertanddevaluingthemother’srole.
Ifthedynamicbetweenphysicianandpatient/advocateistobetruly
collaborative,theconsciousdecisionbydoctorstopositionsthemselvesand
patients/advocatesasequalpartnersinthemedicalprocessisparamount.Sucha
perspectiveisinoppositiontothepositioningofthephysicianas“expert”,withparents
passivelyreceivinginformation(Elwynetal.,2012).Myfindingsrevealthatincases
whereparents’concernswereinvalidatedbyphysiciansduringtheinitialphasesoftheir
children’smedicalcare,physiciansoftenpositionedthemselvesasexpertsunwillingto
considerparents’perspectives.Suchisdemonstratedinthefollowingexcerptfromthe
interviewwithParticipant3.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
46
Participant3:BecauseeverytimeasamomI’vecomeinandsaid,I’vetoldthem
whatIthinkandIdon’tthinktheywantamomgoingontheinternetormaking
theirownjudgmentcallsanduh,Idon’tknow.
Interviewer:Somaybethecultureofmedicineofthey’retheexperts…
Participant3:Yeah,Ithinkso.Likethatonedoctorthatwegotintoabig
argumentwith.Iwassomadathim.Isaidlike,“Youseeherfor2minutes.Isee
hereveryday.Andhowdareyouthinkyouknowherbetterthanme?”Like,even
iflikeyouknow,shewasthehappiest,healthiest-lookingkid,likefora2-minute
blip,it’snotareflectionoftheentireday,youknow?Andit’sjustalsomother’s
intuition,Ithinkaswell.Bigtime.Theydon’ttakethatseriously.Andthatisvalid.
Forsomeoftheparticipants,consultationswithfamilyphysicianscomprisedof
invalidatingtheparents’experiencesoftheirchildren’s’healthaswellasbehavingina
mannerunreceptivetotheparents’concerns.
Alwaysonthelookout:Doingeverythingpossibletomaintainthechild’s
health.Whendiscussingtheirexperiencesinevaluatingtheirchild’sdiagnosesand
processingtheirrelatedemotions,themajorityofparentsinterviewedindicatedmedical
follow-upsasplayinganintegralroleintheirattitudetowardtheirchild’scondition.
Simplyhavingtheirchildassessedbyaphysicianyearlyaffordedparentsthe
opportunitytoreframetheirstress,createspaceforhope,andevenperceivetheir
experiencewithinthemedicalsystemas“positive”.Overall,yearlymedicalexaminations
bytheirchild’sphysiciansallowedtheparentstoperceivethenatureoftheirchild’s
conditionstobelessthreateninganduncertain.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
47
Participant1,whosewifehadpassedawayasaresultofcomplicationsofJuvenile
PolyposisSyndrome,expressedthereliefhefeltinknowinghischildrenwerebeing
medicallyfollowed.Heshared:
Çaabienétéaveclemédecin.Ilaconfirméqu’ilsavaientlepolypose.Mafilles’est
suivie.Ilsl’ontpoignéjeune,ilsontditqu’ilsavaientbeaucoupderisqueque
s’aggravepuisquequ’ilssontpassuivis…Lemomentqu’onsait,onporteplus
attention.[Lemédicine]etsonéquipe,ellem’avaitditqueçan’arriverapas
commeçaparquec’estmoinssurveiller.Elleaditquec’estàdateetc’estclair
qu’estcequienait.J’espèrequeça….contreça.Onsaitqu’ilssontsuivietqu’ils
s’occupentlesaffairesetc’estbeaucoupmoinsstressant.
Participant5explainedthatforher,herchild’smedicalappointmentsalso
providereassurance.Shestated:
Um,and[thedoctor]herselfhas,Ithink,madethisexperienceverypositivefor
us,inthesensethatuh,youknow,shealwaysreassuredusandsaid“He’sonour
radarscreenandbetterwemonitorhimandifsomethingturnsup,we’llcatchit
earlyandifnot,youknow,noharmdonekindofthing”.Hewillhavebeen
followedintohisadultyears.
Furthermore,theconsistencywithwhichappointmentsarescheduledallows
parentstochallengetheiranxietyanduncertaintyconcerningtheirchild’sconditionby
meetingwiththeirchild’sphysiciananddiscussingtheprogressionoftheirchild’s
condition.Suchregularityofconsultationaffordsparentstheopportunitytobuildtheir
confidenceandhopeintheirchild’sfuturehealthaswellasplanaccordinglywiththe
physician.Participant3stated:
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
48
Yeah,sothemysteryhasbeentakenoutofitabit.Weknowwhatwe’redealing
with.We’reseeingaslightpattern.Andwe’reseeingadigression,whichisgreat.
Sothingsarepositive.SoI’mbeingpositivewiththat,youknow?
Themedicalfollow-upswereseenasintegralinmaintainingthechildren’shealth,
bothcurrentandfuture.Furthermore,themedicalvisitsaidparentstoeasetheirfearsof
thedevelopmentofcancerintheirchildren,athemethatwillbediscussedfurtherinthe
remainderofthissection.
Whendiscussingtheirchild’sphysicians’recommendationsthattheirchildbe
medicallyfollowedwithregularfollow-ups,allparticipantsexpressedtheresponsibility
theyfeelforensuringtheirchildrenattendallfollow-ups,scheduledendoscopies.
Participant3explainedthatforher,ensuringherchildattendsallmedicalfollowisher
bestwayofensuringthatherchild’sconditiondoesnotprogress.Sheexplained:
Yeah!AndjustbecauseIfeellikeifweweren’tonit,shecould,right?Butwe’reon
it.Soyouknowandtheyassureusallthetime,‘Thesearenevercancerous,these
arenevercancerous’.So,Ibelievethem.Andthatisonething,youknow,thatit’s
notevenanissue,it’snotevendiscussed,it’ssortoflike,ohnonono.But
obviouslyitcouldbeatsomepointbutIamundertheunderstandingandIam
firmlyunderthebelief,andthisiswhatI’vereadandI’veheardfromthem,thatit
isonlywhentheyareleftuntreated…soIamgoingtomakesurethattheyare
neverleftuntreated.AndIguesswhentheyareadultsandIcan’tdragthemtothe
hospital,well,it’sgoingtobeadifferentstory.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
49
Incommunicatingtheimportanceofmedicalvigilance,follow-upsfortheir
children,physiciansofferparentstheopportunitytoactivelycopewiththeuncertainty
andanxietyresultingfromthepolyposisdiagnosis.Participant7stated:
Oui,pournousleplus…lequoiquiestlepluslourdlàdans,c’estdefairepasserà
[monfils]lescolonoscopiesàchaqueannéeouà,enfinplusieursfois.Donc,ça
évidemmentc’estunprocessusquiestunpeuinquiétantmaisquiestnécessaire.
Alors,pourrépondreàlaquestion,ons’estadaptédanslesensqueoui,onsavait
dequoiils’agissait,onsavaitégalementaprèschaqueexamenqu’elleétaitla
situation.Donc,onpouvaitgérerleportraitavecça.Alors,jediraiquelefacteurle
plusimportantcomedanstoutescesquestions,c’estlacommunication
d’informationpertinents,lapartdesmédecins,desinfirmièresauxparents.Et
sansinformationsadéquates,c’estcertainquelesparentsvonts’inquiéterouils
vontpaniquer,qu’ilsvontsupposertoutessortesdechoses,parcequec’estla
naturehumaine.Maissionlesoffredesrenseignementsprécisetàjour,etqu’on
faitdefaçonrégulière,jesuisconvaincuqueçavacalmerunegrandepartiedes
inquiétudesdesparents.
InthecaseofParticipant4,medicalsupervisionandfollow-upsforherchild’s
conditionservedasanadequatecourseofactiontothesituationanddecreasedthelevel
uncertaintyandanxietyrelatedtothediagnosis.Sheexplained:
Yes,that’strueandsotheotherthingisthatIhaveafriendwhoisadoctor.He’s
retired,quiteelderlynowbuthehadsaidtooatthattimewhenItoldhimabout
it,hesaid‘Oh,ifyoustayontopofit,itshouldn’tbeaproblem’.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
50
Whilemanyparentsdiscussedtheanxietyassociatedwithpreparingtheir
childrenfortheirroutineendoscopiesandawaitingtheirresults,allparticipantsvoiced
theimportancetheyplaceonensuringtheirchildrenaremedicallyfollowed.When
askedwhatadvicetheywouldsharewithotherparentsofchildrendiagnosedpolyposis
conditions,manyadvisedthattheyensurethattheirchildreceivesthepropermedical
care.Participant1cautioned:
D’yfairesuivres.Denepasmanquez[lessuivis],desfoisçachangevite.
Feelinggrateful:Itcouldbeworse.Whendescribingtheirexperiences,themajorityof
participantsexpressedgratitudeasawayofframingtheirexperiences,regardlessofthe
levelofseverityoftheirchildren’sconditions.Theparticipantsdescribedtwofactorsfor
whichtheyaregrateful:thefactthattheirchild’sconditionismanageableandnot
“worse”,andtheopportunitytobeawareoftheirchild’sconditionandtobeableto
remainvigilantoftheirchild’shealth.
Parentsoftendescribedfeelinggratefulfortheirchild’scondition,asit“couldbe
worse”.Participant7explainedthatforher,focusingonherandherchild’sabilityto
managehisconditionwassomethingtobeappreciated.Sheexplained:
That’showyouhavetomakeitthrough;youknowyoucandealwithit.
Participant5explainedthatherchild’sdiagnosisencouragedhertoreflectonthe
importanceofhealth,explaining:
Well,itcertainlyforcesyoutopauseandtoreflectonthetruevalueofhealthand
beinghealthy.Andum,soIcertainlydon’ttakeanyofthatforgrantedanymore…
Imean,Idon’tknow,gratitudeisthefoundationofeverything,youknow?Imean,
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
51
um,Ifeelsoblessed.Youknow,Isupposethediagnosisforoursoncouldhave
beenfarworseanduhwecouldhavebeeninacountrywhereallofthis
healthcareisnotaccessibletous.WecouldhavebeenintheUSwhereitcould
havecostusthousandsofdollars;ithasn’t.[Thiscity]hasthisamazingphysician
in[doctor].Um,yeah,Imeanit’sfantastic.
Inlinewithfeelinggratefulfortheirchildren’shealth,theparentsalsoindicated
gratitudefortheopportunitiesaffordedtotheminordertobeabletomaintaintheir
children’shealth.Suchgratitudehelpstheparentstobeabletoreframetheirchildren’s
difficultmedicalproceduresrelatingtotheirpolyposisconditions,andmitigatetheir
experiencesofworry.Whendescribingtheroleofgratitudeinobtainingmedicalcare
forherchild,Participant4explained:
Imean,you’realwayskindof,notworried,worriedissuchastrongword,butyou
kindofthinkumm‘hey,mykidisgonnagoundergeneralanaestheticagain,that’s
notnormal.That’snotanormalthingforpeopletodo.’But,againthankfulthat
wecandoit,right?Thisisawaythatwecanmaintainmydaughter’shealth,so
it’sokay,itdoesn’tbotherustoomuch.
Additionally,participantsexplainedthateveryprocedurethatrevealspositive
resultsprovidesthemwithanotheropportunitytofeelgrateful.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
52
Figure2.VisualrepresentationofthePeekingthroughcoveredeyesatillnessthemeandsub-
themes.
Peekingthroughcoveredeyesattheillnessliterature
Duringtheinterviewprocess,theparticipantsrevealedtheirexperiencesofseeking
informationregardingtheirchildren’sconditions.ConsistentwiththeComingTogether
fortheWellbeingoftheChildtheme,theresultsindicatethatparentssoughthuman
connection,supportinbuildingtheirknowledge,copingwithuncertaintyandfear
concerningtheirchildren’sfuturehealthtobeimportantintheiradaptationtotheirnew
reality.Thesub-themescomprisingthe‘Peekingthroughcoveredeyesattheillness
literature’themeinclude:a.Seekingvalidationinsharedexperiences;b.Tuningout
anxiety-inducinginformation,whichisdepictedinFigure2.
SeekingValidationinSharedExperiences.Participantsdescribedvalidationof
theirexperiencesasanimportantcopingmechanismandlearningtoolintheirlived
experiencesofparentingachildwithapolyposiscondition.Inadditiontoseeking
informationabouttheirchildren’sconditionsonline,afewparticipantsdiscussed
Peekingthroughcoveredeyesattheillnessliterature
Tuningoutanxiety-inducinginformation
Seekingvalidationinsharedexperiences
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
53
buildingtheirownbankofknowledgebymeetingwithotherparentsinsimilar
situations.Theparentsdiscussedseekingoutotherparentswhosechildrenwere
diagnosedwithpolyposisconditionsinordertolearnaboutthelivesofothers’children,
bothmedicallyandexperientially.Participant3describedconnectingwithaparent
livinginadifferentcountry,andcomparingherchild’ssymptomsandmedical
experiencewiththoseoftheotherparent.Themotherexplainedthatthroughthisnewly
formedfriendship,shewasabletolearnaboutvariousproceduresandresources
availabletochildrenwithpolyps,andexplorepatternsbetweenthetwochildren’s
developmentoftheconditioninordertobestcareforherchild.Themotherdescribed
herconversationswiththeothermotherasmore“technical”innature,explaining:
I’mmorelookingtolearn.I’mverylike,I’mprettytechnical.Iwanttoknow
exactly,likeyouknow,dotheyhavethesamevitamindeficiencies,dotheyhave
thesametendencies?Andwhatdoyouseewhentheyhitpuberty?Whatdoyou
seemoving,likeallthesesortsofthings.Infact,Ishouldprobablyjuststartablog.
[Laughs]Ireallyshould.
Themotheralsodiscussedbeingveryawareofthelackofinformationavailable
onthetopicofpolyposisconditions,andfeelingthatconnectingfamilieswithsimilar
conditionsmightbeveryhelpfulincreatingaforumtoshareexperiencesofthe
conditions.Withregardstoherself,sheexplained:
Iknow.BecauseIhave,IthinkIhavedonemorethantheaverageperson.Ireally
have.Ihavedonethistodeath.AndIhaveexploreditandresearcheditand
everything.SoIdothinkIhavesomethingtooffer.IreallydothinkIcouldteach
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
54
someonesomethingorIcouldlearnsomethingorwhatever.I’mallaboutpiecing
thistogether.
Inadditiontosharingtechnical,medicalinformation,Participant7foundthat
meetingwitholderindividualswiththesameconditionashersonenabledhertobuild
hopeforherson’sfuture,astheindividualsthatshemetledhappyandsuccessfullives.
Theparticipantdescribedherexperienceofmeetinganindividuallivinga
gastrointestinalconditionassuch:
Shehasnobowelleftbutshesurvives,shelooksgreat,Idon’tknowwhatmeds,I
didn’twanttogetintoherdetailsbutshehadspecialsurgeriesandshe’san
advocate…ButIguesswhatIlearnedis,youknow,doanyofthemfeelsorryfor
themselves?Nope,they’recitizensandtheyworkhardandthey…Sothere’sa
positivethatcomesoutofthatandItry,Iwishmysonwouldhavecomewithme
totheeventatleastbecausehewouldhaveseenthatintheworld,he’snotalone.
Andthat’swhatIwantforhim,Iwanthimtobeabletogrowup,haveanormal
lifeandnothavetostoplivingbecauseofthisdisease.
Learningabouttheexperiencesofotherslivingwithpolyposisconditionsallowed
theparticipanttoobserveotherindividuals’adaptivecopingexperienceswithpolyposis
conditions,mitigatingtheuncertaintyandanxietyofwhatthefutureholdsforherson.
Therarityoftheconditionshindersbothmedicalinformationavailableonlineaswellas
theopportunityfordiscussionaboutparents’experiences.Twoparticipantsinparticular
discussedwantingtosetupadiscussiongroupforparentsofchildrenwithpolyposis
conditions.Participant2explained:
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
55
Somaybeitwouldbehelpfuljusttocomparenoteswithanotherparentandsee
whatthey’regoingthrough,seewhatthey’veexperienced,youknow,what’s
helpedtheirkids,becausethere’snotalotofteenagerswiththis,oryoungkids.
Participant3describedherexperienceinaskingherchild’sgastroenterologistto
givehercontactinformationtootherparentsofpatientssothattheycouldsharetheir
experienceswithoneanother.However,theparticipantstatednothingcameofher
requests,likelyduetoprivacyandconfidentialityissues,andthatsheisthereforeonher
ownintermsofconnectingwithotherparentswithsimilarexperiences.
Tuningoutanxiety-inducinginformation.Virtuallyalloftheparticipants
indicatedthattheyconductedInternetresearchontheirchild’sconditionfollowingthe
diagnosis.Theydescribedlivedexperiencesofanxietyanduncertaintyoftheirchild’s
futurehealthwhenseekinginformationaboutpolyposisconditions.Participant5
describeddoingsoasaresultof“humannature”,as“weallhavethistendency,the
minutewehavesomething,weGoogleit,right?Wewanttoknow”.Participant2
explainedthistendencyasnatural,as“everyone’sprimarytoolforeverythingnowadays
istheinternet”.
WhenconductingtheirInternetresearchconcerningtreatments,lifestylehabits
andprognoses,theparticipantsampleasawholedescribeddisappointingresults.Upon
engagingintheresearch,twoparticipantsdescribedexperiencingdifficultyinfinding
informationregardingpolyposisconditions,especiallyforyoungerchildren.Participant
3stated:
It’sbrutal.There’snothing.Imeaneverythingisaimedat50-year-olds,for
starters.ImeanIcan’tevengetanyadvice.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
56
Participant2alsonotedthe“limited”natureoftheinformationavailableonthe
Internet.Thesameparticipantalsodiscussedtheimportanceofexaminingtheavailable
literaturethroughacriticallens,lesttheavailableinformationbedetrimental.She
explained:
Soyouhavetobecarefulaboutwhat,youknow…Luckily,formydaughter,
becauseIwasgoingtobeapsychologistatonetimeandmedicaltrainingandthe
statisticaltrainingthatIreceivedlet’smesortoflookatthingsandanalyzethings
fromaclinicalpointofviewandsortof“Yes,that’sgreatbutwhere’sthe
statisticalbackupforwhatyou’resaying?”,“Whereisthestudies,whereisthis,
whereisthat?”SoI’mnotlikelytofallforwhatevergobbledygooktheyputonline.
Overwhelmingly,parentsdiscussedtheirexperiencesofresearchingtheir
children’sconditionsasanxietyinducing.Readingaboutotherindividuals’“horrible
stories”wasparticularlydistressingtotheparents.Participant4describedher
experienceinresearchingherchild’sconditionassuch:
I’mnotaninternet-reader,Idon’tliketofreakmyselfoutsoItendtostayaway
fromtheinternet.SoIdon’tneedto,Idon’tneedtogooutandlookforallthe
horriblestoriesbecauseit’snotgonnahelpus.
Participant2echoedthissentiment,stating:
See,Iwouldn’tbeabletotuneoutthehorriblethingsthattheysaythey’regoing
through.
Asaresultoftheemotionselicitedbytheironlineresearch,parentstendedto
consciouslyavoidseekinginformationfromtheInternetbutrather,placedfurthertrust
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
57
intheirchildren’sphysiciansforinformativedialogueabouttheirchild’scondition.
Participant5explained:
Uhno,Ithinkit’sasmytrustin[mychild’sphysician]grew,thatumIrealizedwe
wereinreallygoodhands.Andalso,ImeanI…Youknow,I’mjustasguiltyasthe
nextperson,youknow?IGoogledaswellandIfoundmyselfgettingworkedupby
youknow,youclickhere,youclickhereandthenyoudogettocoloncancer,you
dogettoworstdiagnosispossible.Andthen,youknow,soonafterIstarted
realizingthis,Ijuststoppedandsaid,youknow,howamIhelpingmyself?Howis
thishelpingmyson?Howisthishelpingus?It’sjustcausingmoreangstinthe
houseandwedon’tneedit.SoIhadtoadoptaverydisciplinedapproachand
makingadecisionthatIwouldn’tGoogleitanymore.Ithastobeaverycognizant
decisionthatonetakesforthebenefitofeveryonearoundyouorelseitcandrive
someonecrazy.
Forthosewhoinitiallyreadaboutothers’distressingexperiences,they
consciouslyremindedthemselvesthatsuchanexperiencewouldnotnecessarilybetheir
child’sfuture.Forexample,Participant2stated:
Butthemajorityofthepeoplethatgoontheseboardsarepeoplethatare
chronicallyaffectedbyit.So,they’relike,“Oh,youknow,Ijusthadanother
surgery”or“Ilostapartofmybowel”,orthisorthatortheotherthing.Andwhy
wouldyoubombardyourselfwithsuchhorrorsonadailybasisandreadabout
thisstuff?[Laughs]Imean,Ifeelforthepeopleandeverythingbutumtheir
experienceisnotgoingtobe[mychild’s]experience.Andmaybeshewon’tlive
past30,maybeshe’llbe90.Wedon’tknow.Wecanonlytakeitonedayatatime.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
58
Asaresultoftheparents’distressingorineffectiveexperiencesseeking
informationonline,theyplacedevengreatertrustintheirchildren’sphysicians,andthe
medicalsysteminitsentirety,ascompetentinformation-providers.Participant6
explainedhistrustinthemedicalsysteminprovidinghimwithinformationconcerning
hisson’shealthasthefollowing:
C’estcertainlorsqu’onlitdansunarticlemédical,dansdesinformations
médicales,lesinformationssontsouventgénériqueougénéralesourédigéspourrefléter
lesituationdanssonensemble.Maisçapeutpasnousinformersurlecorpsprécisde
notreenfantparcequelaconditionestunpeudifférente,lessymptômespeuventêtreun
peutdifférentes.Enfin,alorsc’estutilepourcomprendrel’ensembleduportraitmaison
aabsolumentbesoinévidemmentdestraitementsmédicaux,desconsultationsmédicales
aveclesmédecinspourbiensaisirqu’estcequiarriveaveclespatientsquinous
concernent.
Participant5,whenaskedhowshewouldadviseotherparentsofchildrenwith
polyposisconditions,respondedwiththefollowing:
Iwouldsay‘Haveconfidencein[yourchild’sphysician].Iwouldsay‘Stayoffthe
internet’.Don’tbe,uh,don’tbeyourselfdiagnosisdoctorforyourchild.Staygrounded.
Don’tletyourimaginationrunwildbecauseitwill.Sononeedtopre-diagnoseanything.
Whendescribingtheirexperiencesoflearningoftheirchildren’spolyposis
conditions,themajorityofparentshighlightedtheirfearofcancerresultingfromthe
polyps.Furthermore,manyexplainedthatthisfearcontinuestobepsychologicallyand
emotionallyimpactful.Whendiscussingthespecificmomenthewasinformedofhis
eldestchild’spolyposisdiagnoses,Participant1stated:
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
59
Ilyavaientdesgrossesdecraintes.Ilsvontdevenircancéreuxauxmoments
donnés.L’espritdisparaitreunpeu.
Forparticipant5,cancerwasattheforefrontofhermindwhenshewasfirsttold
ofherchild’scondition.Sheexplained:
Well,Ithinkit’sinstant.Cancercomesintoyourmindinstantaneously.Whether
myfather-in-lawhadpassedaway[ofcoloncancer]ornot,Ithink,was
immaterial.Themomentthatyoursonoryourchildgoesthroughanendoscopy
andcolonoscopyandthedoctorsays,“I’veremovedpolyps”,regardlessofthe
size,thefirstinstant,youwonderifthey’recancerous.Instant.Imean,thatwas
myreaction.
Shefurtherexplainedheremotionalreactiontothepossibilityofherchildhaving
cancer,herreferencetotheconditionas“theC-word”conveyingthefearshe
experienced:
Um,therewasinitialconcernbecausemyfather-in-lawdiedofcoloncancer,so
youknow,wealwaysdreadedthatC-word.Ithinktherewasalotofconcern
aboutthat…Andthentheyweresentofftothelabtogettested.Andwewere
fraughtwithanxietyandstress.Imean,hewasababy.Atleast,inourminds,he
wasababy.Defenseless.So,thatwasa,youknow,toughtimebutyouknow,we
kept,westayedpositiveandfocusedonthegoodandintheend,youknow,we
wereveryfortunatethattheresultscamebacknegativeandourchildis14now
andhe’sthrivingandsowe’relucky,we’reverylucky.
Withthosewhocontinuetofearthedevelopmentofcancerintheirchildren,
uncertaintyandthechronicnatureofpolyposisconditionswerefoundtoplay
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
60
contributingrolestotheiranxiety.Participant1explainedthefactthatheisacutely
awarethatalthoughhischildrenhavenotyetdevelopedcancerouspolyps,the
possibilityofsuchadevelopmentexists.Heexplained:
Participant:Surtoutlesémotions,quec’estduràcontrôler…Tusaisqu’ilssont
poignéàvieavecca.Mêmesic’estpascancéreux,c’esttropstressant.Les
émotions,etilsremontentvites.
Interviewer:Etpourvous,lecancerestencore…
Participant1:…plusprésent,hein?Canerestepastropdanslepassé,hein?Même
s’ilsn’ontpaseujusqu’àdate,caneveutpasdirequ’ilsn’aurontpas.Mêmes’ils
ressortentlàcomme[lemédecin]adit,c’esttrèsrarequ’ilsviennentcancéreuses.
Theuncertaintyofhowtheconditionsoftheirchildrenwillprogress,forsome,
laststhedurationofthecondition.Participant7explained,inthefollowingexcerpt,that
foraslongashersoncontinuestolivewithJuvenilePolyposisSyndrome,shewilllive
withtheuncertaintyandfearofcancerdevelopmentinherson.
Interviewer:Was,againthismightbeasensitivetopic,butwascancerevera
concernforyou,orafear?Canyoutellmeaboutthat?
Participant:Oh,yes,absolutely.Always.Imeanthatisfrom,youknow,theytell
you‘We’renotsure,we’renotsure,we’restillchecking.We’renotsure’.Well
that’sthefirstthingthatyou’regoingtothink.Orwillitturnintothat?Isitgoing
tobecoloncancer?Idon’tknow.I’llbethinkingaboutthatuntilI’mdead.
InthecaseofParticipant3,suchfearofcancerandtheanxietysurroundingthe
possibilityofitsdevelopmentfosteredfurtheranxietyconcerningherdaughter’soverall
healthandmistrustofthedoctors’viewpoint.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
61
Like,Ikeepsaying,youknow,“Isthereanychanceshecouldhaveapolyp
anywhereelseinherbodythatifleftuntreatedcouldturntocancer?”Like,Iwant
toknowthis,eventhoughIknowyoudon’tknowforsure,isthatstilla
possibility,youknow?“No,no,no,no,no”But,Idon’tknowifIbelieveit.
Whendiscussingtheirexperienceswithcopingwiththepossibilityoftheir
childrendevelopingcancer,participantsacknowledgedboththedifficultyofemotions
presentaswellastheconsciousdecisiontopersistthroughtheirfear.Participant5
explained:
Iwouldbesurprisediftherewereparentsouttherewhotellyouthattheydon’t
thinkaboutit.It’slikeeverywhere,it’sprevalentbutthenit’showyoumanage
thoseemotionsandthoughtsafterwards.
Whenaskedhowshecopeswithherfearofcancer,Participant7stated:
Idon’tknow.Ijustdo.Youjustdo,yeah.Youdoasmuchasyoucan,ImeanI’m
notsayingthatIjusthavemytimeswhenIdon’t,Iwannahideundermybedand
nevercomeout.Butyoujusthaveto,youhavetoforthesakeofthekids…No,but
quittingisnotanoption.
Figure3.VisualrepresentationofthePerseveringuntilneedsaremetthemeandsub-themes.
Perserveringuntilneedsaremet
Teachingchildrentospeakforthemselves
Trustingmaternalintuition
Feelinggrateful:Itcouldbeworse
Takingitonedayattimebecausethefutureisuncertain
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
62
Perseveringuntilneedsaremet
Parentsdiscussedadvocatingfortheirchildren’sneeds,bothwithinandoutside
ofthemedicalsphere,asanimportantpartoftheirlivedexperiences.Theydiscussedthe
importanceofadvocatingfortheirchildren’sneedsbecauseoftheinherentvulnerability
associatedwithpediatricpatients,theresponsibilitythey’vetakenoninmodelling
advocatingbehaviours,theimportanceoftheirchildrenadoptingadvocatingbehaviours
duetothechronicnatureoftheirconditions,andtheroleofmaternalintuitionintheir
advocatingbehaviours.Figure3depictstherelationshipsbetweenthe‘Perseveringuntil
needsaremet’themeanditscomprisingthemes(a.Teachingchildrentospeakfor
themselves;b.Trustingmaternalintuition;c.Feelinggrateful:Icouldbeworse).
Teachingthechildrentospeakforthemselves.Overwhelmingly,parents
discussedadvocatingfortheirchildren’shealthandpreparingtheirchildrentoadvocate
forthemselvestobeanimportantprocesswithintheirparentingexperience.Integralin
theparents’advocatingfortheirchildren’shealthwastheconscioustrainingoftheir
childrentobecometheirownadvocates.Participantsinthisstudydiscussedthatwhile
theycontinuetoadvocatefortheirchildrenbothinsideandoutsideofthemedical
system,theyarebeginningtotakeastepbackinorderfortheirchildrentobeableto
slowlytransitionintobecomingtheirownbiggestadvocates.Itisworthnotingthatthe
childreninquestionare17and19yearsofage,andassuch,fallwithindevelopmental
stagesinwhichindividualsplayanactiveroleinformulatingidentityandaretasked
withtakingongreaterresponsibility(Munley1977).Theparentsnotedthedifficultyin
actingasan“observer…notsayinganything”,ratherthanactingwithintheirgeneral
advocateroles.Oneparentstatedthatherchild’sphysiciantoldthemotherthatherchild
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
63
hadto“speakforhimself”,increasingtheimportanceofdevelopingherson’sadvocating
skillsinherownmind.Thetransitionofpediatricpatients’rolesassimplypatientsto
thatofpatient-advocatesimpelsparentstotransitionfromprotector-advocatetosimply
secondaryadvocate.
Inadditiontothenecessityofparentsadvocatingfortheirchildren’smedical
needs,participantswithinthestudydescribedtakingontheresponsibilityofadvocating
fortheirchildren’sneedsoutsideofthemedicalsphere.Inparticular,participantsnoted
educationalsettingsassitesinwhichtheirchildren’shealthposedproblems.Someofthe
mothersexplainedthatbecausetheirchildren’ssymptomswerenotvisibleandnot
easilyunderstoodbyteachers,itbecameanecessitytoreachouttotheirchildren’s
teacherstoexplaintheirchildren’sconditions,requestflexibilityintheirchildren’sstudy
plansandevaluationmethodsandexplainhowtoproceediftheirchildrenbecameillin
school.Participant2explainedthatshebelievesthattheresponseoftheteacherstoher
child’sconditionanditsaffectonherschoolperformancehaveaffectedherchildona
psychologicallevel,stating:
Anyways,soumalotofthemtheydon’t,they’renotverysympatheticandshe’s
averysensitivepersonsowhenshesensesthatthey’renotbelievingherorthey
startgivinghercrapfornotshowinguporfallingbehind,thenshegetsanxious
aboutitandthenshejustdoesn’twanttogo.
Inordertoprovideherchildwithaneducationalsettingthatwouldprovideher
daughterwithamoreflexibleplantoaccommodatethechild’ssymptoms,theparticipant
researchedalternativeschoolsforherdaughter.However,theparticipantfoundthat
veryfeweducation-relatedresourceswereavailabletoherdaughter,explaining:
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
64
Theydon’tprovidealotofonlinecourses.Ifsomeoneischronicallysicklikeshe
is,thenthereisnoaccommodationforthem.
Assuch,themotherhasfoundittobe“astruggletogethereducated”.Participant
7foundthatherchild’sabilitytoworkapart-timejobwasalsoimpactedbyhis
condition,ashisemployerdidnotallowtheindividualtousethebathroomasneeded.
Themotherdescribedtheabilitytogotothebathroomasonepleasesasoneofthe
“reallyridiculousthingsthatyoutakeforgranted”.Shetookontheresponsibilityof
coachinghersonthroughtheprocessofputtinghisneedsfirst,stating:
AndsoIsaid,“Youdon’tneedthestress,[nameofson].Thestressisjustgonna
killyou,soyouknowwhat?Tellthemyou’redone.Thinkaboutwhatyouwantto
dowithyourself.Getyourselfhealthy.Andsothat’swherehe’satnow.”
Lastly,Participant3explainedtheneedtoadvocateforherchild’sneedstothe
individualssheencountersonadailybasis.Themotherexplainedthatherchildpresents
asveryhyperactive.Theparticipantexplainedthatsuchbehaviourspromptindividuals
whodidnotknowherdaughterwelltojudgeherandherparentingdecisions.She
explained,
Andnoonewouldgivethatkidabreak.IsweartoGod,peoplewouldjustroll
theireyesandI’dsaytothem,like,“She’sanemic.Itaffectsher…It’sbeyondher
control.She’sbeenthroughalot…Theydon’tunderstandthatshe’sbeenthrough
hell.AndI’mnotlettinghergetawaywithmurder.I’munderstandingwhatshe’s
beenthrough,youknow?
Theparticipantexplainedthatshehashadtoexplainherdaughter’sconditionto
othersinorderforherdaughtertobetreatedinawaythatthemotherfeelsisfair.She
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
65
statedthatshefeelsthatotherssometimeshavenocompassionbecauseoftheirlackof
understandingofwhatherdaughterhasgonethroughandthatshehas“always
defendedher”.
Trustingmaternalintuition.Mothersinthestudydescribedmaternalintuition
asplayinganimportantpartinshapingtheirlivedexperiences.Whendelvingintothe
importanceofadvocatingfortheirchildren’sneeds,femaleparticipantscitedtheir
“mother’sintuition”or“gutinstinct”asbeingoneofthedrivingforcesbehindtakinga
standagainsttheirdoctors’invalidationoftheirconcerns.Participant2stated:
Ijusttellanyone,youknowanyofmyfriends,thattheysaysomethingtome
abouttheirkid,Isay,“Well,gowithyourgutinstinctandifyoufeellike,you
know,doctorsarenotpayingattentiontoyouandyoufeelthere’ssomething
morethere,diginyourheelsandjustgoandthrowamummytantrumonthem
untilsomeonepaysattention.”
Theparticipantsexplainedthattheyviewedtheirmaternalinstinctorgutinstinct
asbeinga“valid”toolintheiraimtoprotecttheirchildren.Fromtheirperspective,their
doctors’invalidationoftheirconcernsand“mother’sintuition”servedasapersonal
affrontonthevalidityoftheirrelationshipwiththeirchildrenandknowledgeoftheir
children’sexperiences,withParticipant3stating:
Likethatonedoctorthatwegotintoabigargumentwith.Iwassomadathim.I
saidlike,“Youseeherfor2minutes.Iseehereveryday.Andhowdareyouthink
youknowherbetterthanme?”Like,eveniflikeyouknow,shewasthehappiest,
healthiest-lookingkid,likefora2-minuteblip,it’snotareflectionoftheentire
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
66
day,youknow?Andit’sjustalsomother’sintuition,Ithinkaswell.Bigtime.They
don’ttakethatseriously.Andthatisvalid.
Inthecasesofthesetwoparticipants,theirgutinstinctsormother’sintuition
allowedthemtorefusetoacceptdoctors’initialincorrectmedicaladvice,asitdidnot
feelrighttothem.Suchexperiencessupportthecallformoredynamic,collaborative
relationshipsbetweenphysicians,patientsandtheiradvocateswhereinsymptomsas
wellasexperiencesarediscussedandviewedasvalid.
Takingitonedayatatimebecausethefutureisuncertain.Whendiscussing
thechronicnatureoftheirchildren’sconditions,manyparentsdiscussedtheuncertainty
oftheprogressionoftheirchildren’sconditionsasdifficulttocopewith.Suchfeelingsof
uncertaintyweredeeplyembeddedwithintheirlivedexperiences,withtheuncertainty
motivatingmanyoftheiradaptivebehavioursandmedicaldecisions.Theyalso
explainedthatwiththeirchildren’schronicillnesscomeschronicstressforthemas
parents,astheyoftenfeelhelplessinalleviatingtheirchildren’spain.Furthermore,the
inabilitytoimaginewhattheirchildren’sfuturehealthmightlooklikeorpredicttheir
child’snextintestinalflareupseemstocompriseanimportantelementintheir
experienceofanxiety.Participant3stated:
Imeanwejustdon’tknowhowthesethingsaregoingtoplayout.Butthat’sthe
thing.ImeanIgetthattheydon’tknoweitherbutyoukindofwishyoucouldlook
forwardwhereyoucango“Ohokay,thisisgoingtobeannon-issueinherlife
becausemaybeit’lljustgoaway”.Buttheproblemis,isthateveniftheygoaway,
likewho’stosayonecan’tjustallofasuddenform?Abigone,andmaybeit’s
reallyfast.Andmaybeifshegetspregnanteven,it’llgrow,youknow?It’salways
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
67
goingtobethismysteryof‘Howfastwilltheygrow?”AndIdon’tknowifthey’ll
ever…God,bythattimetheyhaveanyrealanswers,whoknows,right?
Participant2explainedthatcopingwithsuchuncertaintyisdifficult,seemingly
impossibleandhascreatedanexperienceofhelplessnessandhopelessnessforher.She
explained:
Imean,it’slikeanightmarethatyoujustdon’twakeupfrom.It’sjust
like…becauseshe’snotwell,becauseit’salmostadailythingwithus,it’sjustlike
constantstress,constantuncertainty,constantlikeyouseeyourkidstruggling
andyoujustwantthemtobebetterandthey’renot.Andthere’snowaytomake
thembetter.Andyoudon’thaveamagicwandandyourkidsortofexpectsyoudo
tomakethingsbetter.Andyoucan’t.Howdoyoudealwiththat?Howdoyou
adapttothat?Youdon’tadapttothat.[Laughs].There’snoadaptingtothat.
Participant7describedtheuncertaintythattranspireswhenparentinganolder
childwithapolyposiscondition,whosementalhealthhasbeenaffectedbyhiscondition.
Sheexplained:
Oh,honestly,yeah.ImeanI’mworriedaboutitgettingworse.I’mworriedabout
himlosingabowel.I’mworriedabouthimcommittingsuicide.Iguessbecausein
mymindwhenIseehimreallyintheblackzonewhenhe’sreallydown,Idon’t,
youdon’tknowwhatthey’rethinking,right?Soyouareconstantlywondering.
Like,therearetimeswhere,youknow,he’sbeeninhisroomtoolongandI’llgoin
thereandI’mafraidwhatI’llsee.So,hecertainlydoesn’twanttoseekany
professionalhelpbecauseheseesnothingwrongwithwhathe’sdoingsoIreally
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
68
trytotalkhimthroughstuffandtrytogethimto,youknow,thereareother
things...
Moreover,assheexplainedinherexperienceinthefollowinginterviewexcerpt,
thechronicityoftheconditionandoftheresultinganxietyshefeelslimitsherabilityto
processheremotions.
Participant7:…Soyou’remoredealingwithtryingtokeephimoutoftheanger
zonethatyoudon’treallyhavetimetothinkofemotion,youknowwhatImean?
BecauseIstillhavetogotoworkeveryday,Istillhavetofunction,Istillhaveto
youknow…Itreally,Idon’tknow,Ican’tevenanswerthatquestion.Emotionally,
it’shard.Ofcourse,it’shardbutIdon’thavetimetobeemotional.Idon’t…
Interviewer:Itseemslikeyoustillhaven’tevenprocessedit.
Participant7:No.
Interestingly,twooftheparticipantscomparedtheirexperiencesoftheir
children’spolyposisconditionstotheirperceptionsofotherparents’experiencesoftheir
children’scancerdiagnoses.Themothersstatedthattheyfeltthatthepossibilityof
remissionofcertainformsofcancerandthenon-chronicnatureofcertaincancersmight
beeasiertocopewiththantheirchildren’spolyposisconditions.Participant2stated:
Itwasjustlike,‘Okay,there’spolypsandthat’swhat’scausingtheinternal
bleeding,sookay,weknowit’snotcancer,phew.’It’snotthatbutyouknow,in
hindsight,cancermighthavebeenabetterthingbecauseyouknow,ifit’snotthat
advanced,youcantreatitandbedonewithitwhereasthisisalifetimesortof
thing,right?It’snotgoingaway.It’snotgoinganywhere.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
69
Participant3describedaconversationshehadwithfellowmothershehadmet
whosesonwasinremissionfromcancerandwhohadinitiallyinvalidatedthe
participant’sdaughter’sdiagnosisofJuvenilePolyposisSyndrome.Sheexplained:
Butit’sfunnybecauseIranintoherafterwardsandIwaslike,youknow,“How’s
[yourson]?”“Oh,he’sfine,totallycleanbillofhealth”andthenshewaslike,“Oh,
how’s[yourdaughter]?”Andthat’swhenIwaslike“Oh,yeah”,sonowyoumight
getit.Like,yoursondoesn’thavecanceranymorebutmydaughterstillhasthis
condition.Sosuddenlywhatwasunimportant–andIjustsaid“Oh,she’sfine”,
whatever–butitwasabitofalightbulbmomentwhenIwaslike,“Yeah,nowthis
seemsslightlymoreimportanttoyou.”
Themajorityofparentsstatedthatremainingvigilantregardingtheirchildren’s
healthandensuringtheirchildrenparticipatedinthemedicalfollow-upswashelpfulto
themincopingwiththeuncertaintyimplicatedintheirchildren’sconditions.
Furthermore,adaptingtheirexpectationsfromhopingtheirchildren’sconditionswould
eventuallybecuredtosimplymanagingtheirchildren’ssymptomswasrevealedtobe
helpfultosomeparticipants.Participant7discussedsettingsmall,achievablegoalsfor
herson’shealth.Sheexplained:
Well,IthinkI’mjust…Idon’tthink…Like,IthinkIjustconstantlyworry.Like,I’m
alwayslookingfor…Youknow,you’reinthebathroomalittlebittoolong,areyou
okay?Justoverthetopnow,right?He’sprettygoodthoughtotellmewhenthings
arereallynotgoingwell.Buttheydon’tgowellalot,soourrelationshipnowisall
about“howdoyoufeel?”Youknow,not“Oh,howwasyourday?Whatdidyoudo
today?”It’sjust,“Didyouhaveagooddaytodayorwasitaregularday?”,you
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
70
know?Soyes,Idon’tknowifthatwilleverchangebecausehisconditionseemsto
besomethinghe’sgoingtohavetolivewith.Ourgoalisnottohavesurgeryagain.
Ourgoalistokeeptheinflammationatbay,right?Sohowdoyoudothat?
BecauseI’mnotgoingtotellhimwhattoeatatnightorwhattodoorwhatnotto
do.I’llhavetodealwithitwhenithappensthough.Iwonderifthey’llletmestay
withhiminthehospital.[Laughs]
Participant6explainedthatwhileheunderstandsthathisson’sconditionis
chronic,hetakessolaceinthefactthattheillnessisundercontrol.Hestated:
Donc,c’estpaspourdirequec’estfinitpourjamaismaissembleêtresous
contrôle.
Figure4.VisualrepresentationoftheCreatinganew‘normal’aseverythingbecomesunsettled
themeandsub-themes
Puttingfamiliestothetest:Copingwiththeillnessasafamily
Creatinganew‘normal’aseverythingbecomesunsettled
Becomingadifferenttypeofparent
Trustingtheywillspeaksotheycanbeheard
Feelingasthoughnooneunderstands
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
71
Creatinganew‘normal’aseverythingbecomesunsettled
Thefinaltheme,Creatinganew‘normal’aseverythingbecomesunsettled,
comprisesthefollowingsub-themes:a.Becomingadifferenttypeofparent,b.Trusting
theywillspeaksotheycanbeheard,c.Puttingfamiliestothetest:copingwiththeillness
asafamily,andd.Feelingasthoughnooneunderstands.Thesub-themes,depictedin
Figure4,expandonhowthepolyposisdiagnosisaffectedthelivedexperiencesof
parentsonarelationallevel.Furthermore,theywilldiscusstheparents’lived
experiencesoftheirevolvingidentitiesandhowsuchchangeshaveinfluencedtheir
relationshipswiththemselvesandothers.Thefindingsrevealthatsuchchangesaffect
thewaysinwhichtheparentsseekandreceivesupport.
Becomingadifferenttypeofparent.Whendiscussingtheirlivedexperiences,
parentsdescribedanevolutionintheirself-identitiesasparents,whichfacilitatedand
motivatedtheiradvocatingandmedicaldecision-making.Overwhelmingly,participants
statedthatoncetheirchildrenwerediagnosedwithpolyposisconditions,theybeganto
viewthemselvesas“protectors”oftheirchildren.Theparticipantsdescribed‘protecting’
theirchildrenfromtheirconditions,butalsonotedprotectingtheirchildren’sinnocence,
mentalhealth,senseofnormalcyandfamilyrelationships.Participant1discussedtrying
tocreateabubbleofprotectionaroundhischildrenwithoutsmotheringtheminhisrole
astheirprotector:
Maisc’estsurquetutevoiscommeprotecteurunpeu…J’essayedelesprotégéun
peupluslà.Essayedefaireunebouleplusautoursanslesétouffer.Tuprovoques
qu’estcequ’ilsfontetcommentçavaàl’écoleett’esplusprocheàeux,tulestiens
proches,tuveuxterapprocherunpeuplus.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
72
Participant7echoedParticipant1’ssentimentsintakingontheroleof
“protector”forherson,althoughsheusedtoterm“TheCaregiver”todescribeher
experienceinparentingherson.Sheexplainedherevolvedroleassuch:
ImeanIsanginabandfor15yearsandIwasalwaysoutanddoingthat.Andthen
IquitbecauseitwasjusttoomuchtooworryabouthimanddoyouknowwhatI
mean?Youjustcan’tdoitallandbeyourself.Youcan’tbewhoyouareandbe
momandbe,youknow,andthenbethecaregiver.IcallitTheCaregiver,Imean
youareaparent,youshouldbebutit’sjustthatextralevelofworrythatyoutake
forgranted.
Themotherexplainedthatanimportantdistinctionbetweenmotherand
caregiveristheexperienceofworryandanxietyshefeelsasherson’scaregiver.Asshe
explained,intheroleofacaretaker,itisnecessarytoplaceherchild’sneedsbeforeher
ownandprioritizetheadoptedroleofcaretakeraboveotheridentitiesshemayhold.
Trustingtheywillspeaksotheycanbeheard.Whendiscussingtheir
relationshipswiththeirchildren,themajorityofparentsdescribedadevelopmentof
trustthroughcommunication.Buildingsucharelationshipwasessentialtotheir
parentingexperienceandplaysameaningfulroleintheirlivedexperiences.They
explainedthattheirchildren’slevelofcommunicationandtrustinthemasparents
servedasanecessity,astheparentsneededtobeawareoftheirchildren’spolyposis-
relatedsymptoms.Participant5describedtheimportanceofcommunicationwithher
childrenassuch:
AndIcommunicatewiththekidsregularlyabouteverythingbecauseifsomething
doesshowupintheirstool,itcannotbeanembarrassmentforthemtotellus.If
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
73
whateverishappeningishappeningtotheirbodies,theyhavetohavethatlevelof
confidenceandcomforttocometous.Soifanything,IthinkI’veprobablybecome
anyevengreatercommunicatorwithmykids.
Thetrustandcommunicationbuiltbetweenparentandchildalsocreates
opportunitiesforparentstoteachtheirchildrenhowtomanagetheirconditionsthereby
removingthestigmaofdiscussinguncomfortabletopicssuchasbloodinstoolandstool
consistency.Participant3describedhercommunicationwithheryoungchildrenabout
theirconditionsasthefollowing:
Andthechildrenaretrainedtolookattheirstool…Nottheolderones.[My
children]arealways,“Ahmom,there’sblood”,youknow.They’resononchalant.
“Isawblood”“Okay,showme”,youknow?Soit’slike,ah[sigh].Anyoneelse
wouldbelike…[Laughs].I’mjustsoblaséaboutitnow.Youknow,andI’lloften
havetochecktheirstool.Isitsolid?Isit,youknow,iftheyhavechronicdiarrhoea,
that’satipoffthatthere’ssomethinggoingon,meaningthatit’sblockingthe
stool,right?
Children’scommunicationoftheirmedicalsymptomstotheirparents,especially
inthecaseofyoungerchildren,facilitatesparents’rolesasadvocateswithinthemedical
systemaswellasoutsideofthemedicalsystemastheyarebetterinformedofwhattheir
childisexperiencing.Inonecase,Participant7explainedhowthetrustsheplacesinher
childtonotifyherwhenheisfeelingunwell,helpshertomitigateheranxietyabouthis
condition.Shestated:
IthinkIjustconstantlyworry.Like,I’malwayslookingfor…Youknow,you’rein
thebathroomalittlebittoolong,areyouokay?Justoverthetopnow,right?He’s
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
74
prettygoodthoughtotellmewhenthingsarereallynotgoingwell.Buttheydon’t
gowellalot,soourrelationshipnowisallabout“howdoyoufeel?”Youknow,
not“Oh,howwasyourday?
Suchlinesofcommunicationonlybecomemoreimportantasthechildgrows
olderandotherfactorssuchaspuberty,peerpressureandneedforindependenceplaya
roleintheparent-childrelationship.Participant2explainedthatasherdaughter
progressesthroughherearlyteenageyears,shehasfoundthattheemotionalimpactof
Peutz-JeghersSyndromehasbeguntobecomeatopicthatismoreoftendiscussedin
theirconversations.Sheexplained,
Sheneedstohaveonepersonthatshecanalwaysgotonomatterwhat,especially
withthedepressionandanxiety…IthinkI’mprobablyclosertomydaughterthan
Iwouldnormallybe.
Whiletheparent-childrelationshipsoffamiliesdealingwithpolyposisconditions
paralleltheparent-childrelationshipsoffamiliesnotaffectedbythecondition,these
findingsrevealthatthedevelopmentoftrustthroughcommunicationisofparamount
importancewhenraisingachildwithachronicillness.
Puttingfamiliestothetest:Copingwiththeillnessasafamily.Althoughthey
broughtforthvaryingchallenges,alltheparticipantsdiscussedtheirchildren’s
conditionsashavinganimpactontheirfamilydynamics.Aspreviouslydiscussed,the
participantsservedasadvocatesfortheirchildren,builtuponafoundationoftrust
betweenchildandparent.Theyexplainedthatastheirchildren’sillnessesprogressed,
theirwaysof‘being’withinthefamilyevolved.Theparticipantsreportedthattheimpact
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
75
ofsuchaclose,uniquebondbetweenthemselvesandtheiraffectedchildaffectedtheir
children’srelationshipswiththeirotherparentsandsiblings.
Participant2explainedthatwhileherdaughterwillaskherforadviceandwhile
theyshareaclosenessbetweenthetwoofthem,herdaughterdoesnotfeelthesame
aboutherfather.Sheexplainedthatbecauseherhusbandholds“thissortofdude
mentalityof‘Suckitupandgetonwithit…andjustdealingwith’,herdaughternolonger
seeksthesupportofherfather.Themotherexplainedthatthiscanbedifficultforher
husband,ashe“resents”theclosenessoftheirrelationship.
Participant7alsoexplainedthatbecauseofherhusband’swayofrespondingto
theirson’sillness,hersondoesnotfeelcomfortableseekingsupportfromhisfather.She
explainedthatshedoesnotfeelhersonreceives“alotofsupportfromhisdadbecause
hisdaddoesn’tknowhowtosupporthimwithoutitbecomeafight.Andhisfatherwould
lovetotellhimwhattodo.”
Theparticipantsalsodescribedtheimpactoftheirchildren’sdiseaseintheway
theirchildrenrelatetotheirsiblings.Participant3explainedthatthechangesshehas
madeinherparentingstyleandherroleinthefamilysinceherchild’sdiagnosishasnot
onlyimpactedherchildwithJuvenilePolyposisSyndromebutherotherchildrenaswell.
Shestated:
Ohyeah,wellit’sdefinitelyputusonadifferentpathforsure.Justeverything,like
theconceptofourfamilyinsomewaysandyeah,Isenseher…Likemyeldest,I
cantellthathetreats[childwithJuvenilePolyposisdiagnosis]differently.Ican
tellthat,youknow,mysecond,mydaughter,mysecondeldest,Idon’tthinkshe
reallygetsit,likeshedoesbutIcanseewithmyoldest,he’sconcernedbutnot
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
76
sayingit,youknow.AndsoIcanseehowitplaysonthemabit.AndthenumI
meanit’sgoingtobesomethingthatisalwaysthere,youknow?Andyeah.
Participant7explainedthatherson’sdiagnosisandexperienceofhiscondition
havehadlongstandingeffectsonherson’srelationshipwithhissister.Shestated:
Andhehasanoldersisterwho,Ithinkshe’salmostresentfulatthispointbecause
shefeelsthateverybodyworriesabout[herbrother],right?Andit’s‘causeitwas,
that’sthewayitwas.Imeanwehadafamilyholidaybookedandwehadtocancel
becausehewasn’tallowedtogoanywhere.Andshewasinher,Ican’teventhink
whatshewouldhavebeenin,hewas13,shewouldhavebeen16or17,Iguess.
So,itwasprobablytheendofschool,Iforget.Itwasanimportantyearforher.
Butwecouldn’t,Ididn’thavetheenergytodoitall,right?Andnowtheydon’t
evenhavearelationshipbecauseheresentsherforbeingresentful,right?It
almostsoundslike…Ithinkheinterpretsitasshe’sbeingveryjealousofhim,
whichsoundslikesheis.Whennowthatshe’s24,shecangetoverit,andshe
wantsarelationship,butheisnotthereyet.Soshelivesin[anothercity],shegoes
to[school].Andit’sgoodthatthey’renottogetherrightnow,butit’ssad.It’ssad
becausetheywereveryclose.
Finally,participantsdescribedtheirchildren’sdiagnosesashavingnegatively
impactedtheirrelationshipswiththeirspousesinitially,butultimatelycreatingan
opportunityandneedtoreflectontheirrelationshipforthesakeoftheirchildren.
Participant5describedtheimpactofherchild’sdiagnosisonhermarriageasthe
following:
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
77
Yeah.Imean,Isupposeitdid.Howcanitnot,right?ButIthinktherewassomany
otherfactsthatimpactedourrelationshipthatthatwasjustoneofmany.Soit
wasoneofmanystressors,whicheventuallyledtothebreakdownofthe
marriage.Iwouldn’tsayitwasitwasthesolestressor,farfromit.Iwouldn’teven
sayitwasthebiggeststressor,farfromit.Ifanythingthough,Ithinkthediagnosis
forcedhisfatherandIjusttostopbickeringandstopthedynamicsofour
relationshipandhaveadifferentrelationshipforthiskid,whichishugely
beneficialtodaybecausehisfatherhasre-married,Ihaveremarried,thiskidhas
lotsofstep-siblings,andeverybodygetsalong,youknow?Wegetalongverywell
withhisfatherandhisnewwife.So,youknow,we’reverylucky.There’snoum,
there’snoacrimonybetweenthefamilies,thechildrenallseemtogetalong,so
we’requitelucky.
Participant3describedasimilarexperience,inthatsheandherhusbandwere
abletoadapttheirrelationshipovertimetocreatehealthierdynamicsinordertobetter
supporttheirchild.Sheexplained:
WellIkindofknowwhattoexpectnow.NowIknowexactlywhattoexpect.AndI
stillgetstressedbutlikeyouknowthelasttimewewentin,[myson]wentinthe
lasttime,Ifelt,likeIdidn’tfeellikeIwashavingafull-onanxietyattack,likeIwas
goingtothrowup.Soit’slikelike,‘okay,I’vecomealongway’.Myhusbandis
prettygood,hejustdrawsandzonesout.Andhe’sjustlike“Youcan’tstress,it’s
beyondyourcontrol”,youknow?ButIwill.AndthenItendtosortofspendmy
energyorgetfrustratedatthelackofsupport.Andthatseemstobeafocusbut
whatever…IguessmyhusbandandIareprobablyclosernow,kindofcoming
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
78
throughontheotherend.Therewasatimewhenourmarriagewasverystrained.
Anduh,I’msurethisplayedabigrole.Forsure.
Insum,themajorityofparticipantsdiscussedtensionarisingwhenindividuals’
emotionalandsocialsupportneedswerenotmetwithinthefamily.Theneeds,
specificallyinthecasesofthechildrenwithJuvenilePolyposisSyndromeandtheir
siblings,paralleleddevelopmentalneedsoftheirhealthypeers.However,thestressors
inflictedbytheconditionsincreasetheseverityandimportanceoftheseneeds.
Feelingasthoughnooneunderstands.Parentsexpressednothavingtheir
emotionalandsocialsupportneedsmetbytheirsupportnetworks,asimpactfuland
concerningpartoftheirlivedexperiences.Participantsbroughtforthawidevarietyof
experiencesofsharingtheirchildren’sdiagnosisandoverallhealthwithfamilymembers
andfriends.Myfindingsrevealseveralfactorsaffectingthequalityofsocialsupportthe
participantsreceived.Themajorityofparticipantsdescribednotreceivingsocialsupport
inlinewiththeirneeds.Participant3explainedthatherfamilydidnotmeether
emotionalneedsduringtheearlyyearsofherdaughter’sdiagnosis.Shestated:
Oh,I’mgoingtogetemotionalaboutit.[Crying]Um,becausewehadreallypoor
supportfromourfamily,thatwhatwasthehardestthing,Ithink.Becausewehad
todoa…Ourfamiliesforsomereasontreatitasthoughitiscompletelynothing.
Theyactlike,“Oh,whatever”,like“Getoverit”.Andwhenwehavetodoum,when
wehavetodouhthefastingandstuff,it’sverystressfulbecausewehaveother
children,especiallywhen[ourchild]wasyounger…Ourparentswerejustnot
thereforusatall.Like,wewerelike,ohmyGod,weweresostressedout.AndI
gotstrepthroat.Ihad[mydaughter]and[myson]goinbacktobackfora
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
79
procedureanditwasthefirsttime[myson]hadgoneinandIwasterrifiedof
whattheyweregoingtofind.Um,luckilyitwasonlyone,butyoudon’tknowuntil
yougoin,right?Andhehadquiteabitofblood.Um,hekeptsayingthathewas
bringingupstuffandthathecouldtastebloodinit.And,soImean,andhewas
little.Anduh,mymom,everytimewouldseeourkidsgoin,because[my
daughter]hasbeenin9timesIthink,[myson]’sbeenintwice,anyway,mymom
wouldconvenientlyleavetownandmymomandmysisterdidn’teven…They
didn’tcallmethedaybefore,whentheyknewthatIwasawreck,theycalledme,
wehadtheprocedureinthemorningIthink,theycalledmelike6:00thenext
night.AndIjustlostitonthem.Ijustsaidlike,likewhatever,it’sover,like…You
know,likesuretheyweren’tdiagnosedwithcancerbutinoureyes,itwas
terrifying.So,thatwasn’tgoodforsure.Um,itwasverystressfulonmyhusband
andI,withoutadoubt.Ithinkwe’vekindofcomeoverit.Theworstwaswhen
theywentbacktoback,thatwashard[Crying].
Whenaskedhowshewouldhavelikedtoherfamilytohaverespondedtoher
needs,themotherexplained:
Ijustwouldhavelovedsomeonetohavecalledandsaid“It’sgoingtobeokay,
don’tworryaboutit”,youknow?...Evenjustthat,orgoodluckoranything.You
know,becausewe’realwaysveryaware,likeit’sscarywhenanyoneisputout.
Thereisthechancethatyoudon’twakeup,youknow?Thereis.Um,soIjust
couldn’tbelievethatuh…Ijustdon’tknow.Ijustdon’t.Ireallydon’tknow.Itwas
tothepointofalmosteye-rolling.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
80
Suchresponsesfromherfamilymembersdissuadedthemotherfromseeking
futuresupport,sheexplained:
Idon’teven;Idon’tthinkIwouldevenbringitupagainreallywiththem.
Themotherfurtherelaboratedthatshefeltherfamilydidnotfullyappreciatethe
seriousnessofthecondition,withverylittleunderstandingofthecondition.Shestated:
Theydon’tthinkit’sabigdealbecausetheylookatitaslike…Theyallsaythe
samething…Likemysister-in-lawsays,‘Well,mymomhasapolyp’andsoandso
hasapolyp,butit’saconditionin50-year-olds,it’scommon.Sotothemit’snota
bigdeal,soit’s‘Iknowtonsofpeoplewithpolypsandtonsofpeoplewhohave
colonoscopiesandgetoverit’.Well,hello.Thisisachild.Andhavetheyhad50
polypsremoved?...Theydon’tseemtothinkit’sabigdeal.They’relike,‘justtake
themout’.AndwhenItalkaboutit,Isortofsaylike…Like,sometimesthey’llsay
thingstomeandit’sliketheydon’tunderstandatallandI’llbe…Like,mysister
waslike,“Oh,Italkedtomyfriendandshesaidyoushouldtakethisbecauseit
makespolypsgoaway”andI’mlike,“No,no,youdon’tunderstand.Like,thisisn’t
likepolypslikegrandpahadorwhatever,youknow?Like,thisisdifferent.”
Participant2echoedsimilarsentiments,reportingthatshefeltothersinherlife
didnotunderstandherchild’scondition.Sheexplained:
Theyjust,theydon’tgetit.Theyhaveneverheardofit,theyjustdon’tknowwhat
thismeansandlikeIsaid,yousay‘polyps’anditsoundssocute,youknow?What
couldapolypdotoyou,youknow?Peoplehaveheardofpolypsandthey’relike
“Wellthatdoesn’tsoundsobad,doesit?Youknow,youremoveitandyou’re
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
81
good.”Butwhenthey’reconstantlygrowingandyoudon’tknowatwhatrate,you
can’tseethemontheoutside…
Participant5’sexperienceofsharingthenewsofherchild’sdiagnosiswithher
familydemonstratedthevalueofconveyinginformationandfosteringunderstanding
aboutthecondition.Shestated:
Uh,no.Ithinkeverybodywasjustkindofjust“Oh,whatisit?Whatdoesthat
mean?Whataretherisks?Whatarethepossiblediagnoses?”Andsoitjustledto
veryfactualconversationsandkeepingpeopleapprisedofwhatwashappening.
Andum,Imeanourfamilieswereverysupportiveandveryunderstanding,and
sowereourfriendsandemployers.So,yeah,Idon’tum,Idon’trecallanything
negativeaboutthateither,justveryfactual.
Partofthereasonwhyothersarelikelytounderestimatetheseriousnessof
polyposisconditionsandill-appraisetheaffectedparties’needforsupport,asexplained
byParticipant3,isthefactthattheconditionoftentimesdoesnotpreventchildrenfrom
presentingdifferentlyfromahealthilydevelopingchild.Thefollowingexcerpt
demonstrateshowtheparticipant’sdaughter’soutwardappearancedoesnotreflectthe
child’soverallexperienceandhealthchallenges:
Interviewer:Andespeciallybecauseyoucan’ttelloutwardly.
Participant3:Exactly.
Interviewer:Andyourdaughterisfullofenergy.She’sprobablythelastperson
you’dassume.
Participant3:Exactly.Ifshewassickly,thenshewouldhave…Buttheydon’t
understandthatbybeinglikethat,it’sstillsickly.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
82
Thedifficultyofindividualstoreconcileillnesswithanoutwardly‘healthy’
appearanceextendsnotonlytothoseunfamiliarwithpolyposisconditionsbutto
physiciansaswell.Myfindingsrevealthatphysiciansoftendiscountedparents’initial
requestsforfurtherexaminationoftheirchildren’ssymptomsoroverallhealth,with
physicianssometimesinvalidatingparentsconcernsbystatingthechildren’ssymptoms
weresimplypartofnormaldevelopment.Experiencessuchasthesedemonstratealack
ofsupportfromphysicians,usheringinexperienceswhere,formanyparents,supportis
notavailable.
Anotherreasonwhyindividuals,includingphysicians,areunlikelytoinitially
understandthecomplexitiesandseriousnessoftheconditionistherarityofdisease.
Participant3explained:
Therarityofitdoesn’tgiveitanyexposure,itdoesn’tgiveitanything.
Whiletherarityoftheconditionhinderseducationaboutthecondition,italso
makesitverydifficultofindividualsaffectedbytheconditiontofindotherswho“getit”.
Whendiscussingwhetherbeingincontactwithotherfamiliesaffectedbypolyposis
conditionswouldbehelpful,Participant2responded:
Andmaybe,like,maybeif…Like,howwouldyoufindanotherparentwith,you
know,akidwithPeutz-Jeghers?Howwouldyouevengoaboutthat?Like,are
thereotherparentsinthe[city]area?Howwouldyouevenlocatethem?Like,
there’sdoctor-patientconfidentialityandyouknow,it’snotlike[thedoctor]is
goingtosay,well,‘Ohthere’slikeoneortwootherpeople’…Yeah,Ithinkit
actuallywouldbehelpful,yeah.Andthenevenformydaughter,maybe,tomeet
someoneelseherownagewiththesamethingandsortofsay“Well,youknow”…
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
83
Noone…Shecanexplaintoherfriendsbutnoonereallyunderstandswhatshe’s
goingthrough.
Inadditiontoothersbeingillinformedaboutpolyposisconditions,Participant3
alsonotedthestigmaattachedtopolyposisconditionsbecausetheyaffecttherectum.
Themotherdiscussedthediscomfortshebelievesothersfeelasapossiblereasonwhy
othersarehesitanttodiscussherchild’scondition.Shestated:
Participant3:It’saweird,itisaweirdthing,right?Becauseit’stodowiththe
bum,right?Sopeopledon’twanttogothere,theydon’twanttotalkaboutthat
area.
Withregardstoparents’experiencesofsocialsupport,itisimportanttodiscuss
parents’psychologicalreadinesstoreceivesupport.Participant2discussednotwanting
otherstoengageherinconversationaboutherdaughter’shealth,possiblyduetoher
expectationthatthesupportwillnotmeetherneeds.Shestated:
Youcansmileand“Oh,everything’sfine”butit’snotandyoujustdon’twanttobe
asked.Umandbecauseitdoesn’truninourfamily,noonereallyunderstands.
Lastly,Participant2discussedhesitatingwhethertotellherfamilymembersof
theirchildren’sconditionsforfearofburdeningthem.Sheexplained:
Atfirst,becauseItoldmyhusbandmymotherhashighbloodpressure,bothsets
ofgrandparentsarestillalive,Isaid‘Iamnottellingmyparentsuntilweknow
exactlywhatthismeansandthenwhenwedoknowexactlywhatthismeans,
thenI’lldecidehowmuchorwhattheyneedtoknow.”Because,um,youknow,
it’sjustupsettingtomymotherandshe’lljustworryalotandthenitjustputsher
bloodpressureup,youknow.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
84
DescriptiveIdentificationofthePhenomenonStructure
Indealingwiththeanxietyanduncertaintybroughtonbytheirchildren’s
diagnoses,ourparticipantsactedproactivelyinworkingtoensuretheirchildren’s
currentandfuturehealth,therebyallayingtheirowndistress.Theirmeansofadaptation
reflecttheuseofrelationship-buildingbehaviourstomeettheseends,includingforming
relationshipswiththeirchildren’sphysicianstoensurepropermedicalcare,
strengtheningrelationshipswiththeiraffectedchildreninordertofacilitateadvocating
ontheirbehalf,evaluationoftheirrelationshipswithfamilymembersinordertobetter
adaptfamilydynamicsandreflectionupontheirrelationshipswiththemselvesinorder
tobetterunderstandtheiridentitiesasparents.
Physicianswhowereabletoeffectivelycommunicateinformationwhilealso
makingparentsfeelheard,servedasvaluablesourcesofsupport,whichmyfindings
indicateparentsofchildrenwithrare,chronicillnessesdesperatelylack.Furthermore,
physicianswhowereabletoearnthetrustofparentsbyengagingtheminfamily-
centeredcarewereabletoeffectivelyconveytotheparentstheimportanceofcontinued
medicalsurveillance,animportantcomponentforthecontinuedmedicalcareof
individualswithchronicillness.Parentsalsodiscussedtheimportanceofcreating
relationshipswithotherindividualsinsituationssimilartotheirs,astherarityand
stigmaoftheirchildren’sconditionscanbeisolating.Positiveexperiencesinwhich
parentsareabletosharetheirconcernsandstorieswithothers,whetherprofessionals
ornon-professionals,aremeaningultoparents,evidencedbythegratitudethey
expressedintheinterviews.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
85
Inparentingtheirchildren,theparentsactedupontheirfeltresponsibilityto
teachtheirchildrentheimportanceofadvocatingfortheirneeds,bothwithinthe
medicalsystemandwithinothercontexts.Theysawthisasanintegralpartoftheir
parentingroleduetothechronicnatureoftheirchildren’sconditions,andthe
importanceofcontinuedmedicalsurveillancetothehealthoftheirchildren.Theyalso
discussedhowtheirrolesasparentsandindividualshavechanged,havingtakenonthe
rolesof‘Protector’and‘Caregiver’,andwithit,andtheevolutionoftheirfamily
dynamics.Specifically,parentsexpressedconsciouslyfosteringeffectivemeansof
communicationandtrustwiththeirdiagnosedchildreninordertobetterunderstand
theirday-to-dayhealthandmoreeffectivelyadvocatefortheirchildren’sneedstothe
physicians.Theusualsocialsupportsystemsoftheparentsoftentimesdidnotpresent
theformsofsupporthelpfultotheparentsintheirtimesofneeds,andassuch,they
reportedfeelingisolatedintheirexperiences.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
86
Chapter5:Discussion
Thischapterpositionsmyfindingswithintheresearchliteratureanddiscusses
theirrelevancetothemedicalandcounsellingdomains.Allofmyrevealedfindingsalign
wellwithcurrentchronicillness,rarediseasesandpolyposisliteratures.The‘Teaching
childrentospeakforthemselves’themeprovedtobethemostnovelofthethemes,in
thatthecurrentresearchliteraturehasnotyetexploredthismatterindepth.
Furthermore,inthischapterIbringforthlimitationsthatshouldbeconsideredwhen
reviewingthisstudy’sfindings.Lastly,myfinalconclusionsregardingthisstudyand
possibleavenuesforfutureresearchbasedonmyfindingsarediscussed.
SummaryofFindings
Myfindingsrevealthatadiagnosisofapolyposisconditionbringswithitagreat
amountoffearanduncertainty.Inordertoadaptandcombatthesefeelings,parents
havedevelopedvarioustoolsforallayingtheirfearsandensuringtheirchildrenremain
healthy.Theparentsdiscussedhowtheyexperiencedgratitudeandreassurancewhen
physiciansworkedcollaborativelywiththemtomaintaintheirchildren’shealth.When
parentsfelttheirchildren’sneedswerenotbeingmetortheirinstinctsabouttheir
children’shealthnotrecognized,theydidnothesitatetoadvocatefortheirchildren.In
ordertocombattheuncertaintytheparentsfeltregardingtheirchildren’sfutureandto
bestpreparetheirchildrentomaintaintheirownhealth,theparentsdiscussedtheir
experiencesinteachingtheirchildrentoadvocateforthemselves.Theparticipantsalso
discussedtheirexperienceinbuildingtrusting,communicativerelationshipswiththeir
children,asitisintegralforthemtobeawareoftheirchildren’shealth.Lastly,parents
discussedtheirparentidentitiesandfamilydynamicsevolvingwiththeprogressionof
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
87
theirchildren’sillnesses,withmostindividualsnotunderstandingtheirexperiences.
Theydiscussedseekingotherswhosharetheirexperiencesinordertoreceivethe
validationdeniedtothembytheindividualswhosimplydonotunderstandtheir
experiences,andtobuildtheirknowledgeabouttheirchildren’sconditions.
DiscussionofFindings
Theaimofthisreseachstudywastogainqualitativeinsightintothelived
experiencesofparentsofchildrenwithpolyposisdiagnoses.Thefindingscompiledin
thisstudyaddtothelimitedresearchliteratureontheexpeirencesofparentsofchildren
withchronic,rareillnessesaswellasthepolyposisliterature.Overall,thefindingsofthis
studyparallelthoseofpriorresearchandcontributenovelinsightonthelived
experienceofthisphenomenon.
Whenreviewingthecollecteddata,oneofthemajorthemesthatemergedwas
parents’needtohavetheirexperiencesandconcernsheardandvalidatedbythose
withintheirsupportnetworks,whichincludesphysicians.Whendiscussingtheir
experiences,allparticipantsdiscussedin-depththeirphysicians’abilitiestosupport
theirfamiliesbylisteningtotheirconcerns,informingthemofhowtobestcarefortheir
children,andsupportingthemthroughtheirprocessofadaptingtotheirchildren’s
diagnosis.Whileparentswitnessedtheirchildrenreceivingmedicalcare,theirfocusof
discussionduringtheinterviewsprioritizedtheirexperiencesofthesupportthey
received(ordidn’treceive)anditsimpactontheirexperience.Parentsalsodiscussed
theirneedtobeabletosharetheirexperienceswithotherparentsofchildrenwith
polyposisconditions,toonceagainfeelheard,havetheirexperiencesvalidated,and
possiblybenefitfromotherindividuals’insight.Ourstudydemonstratestheimportance
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
88
ofparentsofchildrenwithrareandchronicconditionshavinganopportunitytoexpress
theirexperiencesandneedsaswellastheimportanceofhearingothers’storiestoknow
thattheyarenotalone.Inthisway,therelationshipsthatarebuiltarehealing.
Thefindingsrevealthatparticipants’relationshipswiththeirchildren’sphysician
playsakeyroleindeterminingthequalityoftheirexperiences.Parents’whose
children’sphysicianstookthetimetoexploretheirconcernsandassesstheirchildren’s
symptomsaccordinglydemonstratedtrustintheirmedicaloverallsystemandexpressed
havingmanyoftheirconcernsassuaged.InastudybyMcCormickandcolleagues(2012),
familyphysicianswerefoundtoinvalidatepatients’experiencesofgastrointestinalpain,
resultinginpatients’emotionaldistress.Aswithmyfindings,Drossman,Chang,Schneck,
Blackman,Norton,andNorton(2009)foundhavingtheirexperiencesandconcerns
understoodandacknowledgedbytheirphysiciansaidedIrritableBowelSyndrome
patients’abilitytofunctionintheirday-to-daylives.Drossman,Creed,Olden,Svedlund,
Toner,andWhitehead(1999)intheirreviewofpsychosocialexperiencesofindividuals
livingwithgastrointestinaldisorderssuggestphysiciansuseapatient-centredapproach
whendiscussingpatients’experiences,exploringbothpsychosocialandphysical
components.Myfindingssuggestthatphysicians’useofpatient-centredpracticeshould
extendtotheparentsofpediatricpatients,aswell,utilizingafamily-centeredapproach.
Asqualityofcarehasbeendemonstratedtoplayasignificantroleinhealth-
relatedqualityoflife,patient-centeredmedicalpracticecontinuestoproveitsvaluein
patient-physicianrelationships(vanderEijketal.,2004).Evenseeminglyunimportant
gesturesthatpatientsinterpretas‘courteous’havebeenfoundtosignificantlybenefit
health-relatedqualityoflife(vanderEijketal.,2004).Theparticipantsexpressedthe
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
89
theimportancetheyplaceintrustingintheirphysicians,withsuchtrustallowingthe
participantstobettercopewiththeiruncertaintyandanxiety.Specifically,myfindings
revealthatacollaborativerelationshipwiththeirchild’sphysicianenablesparentsto
playamoreactiveroleintheirchildren’smedicalcare,therebyaffordingparentsan
opportunitytoactivelycopewiththeirfeelingsofhelplessness.Trustdemonstratedby
patientsintheirphysicianhasbeenshowntobeapredictorofadherencetomedical
practices,animportantfactorincancer-relatedconditionssuchaspolyposis(Nguyen,
LaVeist,Harris,Datta,Bayless,&Brant,2009).Takentogether,myfindingsindicatethat
whileconsultingtheirchildren’sphysiciansandreflectingontheirchildren’sconditions
canbeaveryanxioustimeforcertainparents,follow-upswiththeirchildren’s
physicians,especiallywhenthedoctorsfacilitateacollaborativerelationship,ultimately
playanimportantfactorinreducingparents’anxiety.Specifically,scheduledfollow-ups
andphysicians’surveillanceallowsparentstosomewhatallaytheirfearsofthe
developmentofcancerandunforeseenmedicalissues.
Someoftheparticipantsreportedwantingtobeabletointeractwithother
individualsinsituationssimilartotheirs,withonlinesupportforumsnotmeetingtheir
currentneedsofgainingsocialsupportandcollectinginformation.Althoughallofthe
participantsreportedusingtheInternettobuildtheirunderstandingoftheirchildren’s
conditions,myfindingsdonotentirelyparallelthoseofpriorresearch.Manyofthe
participantscitedinformationfoundonlinetobetoogeneral(andthereforenot
necessarilyrelevanttotheirchildren’sspecificsituation)ortooanxietyinducingfor
themtocontinuetousetheInternetasasourceofinformation.Whileresearch
demonstrateslinksbetweeninflammatoryboweldiseasesandanxiety,thefindings
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
90
provideinsightintospecifictriggersforsuchanxiety(Graff,Walker,&Berstein,2009).In
linewithpriorresearchinstudiesfocusedoninflammatoryboweldiseasepatients,the
participantsreportedviewingtheirchildren’sphysiciansastheirpreferredsourceof
information(Bernstein,Promislow,Carr,Rawsthorne,Walker,&Bernstein,2011;Cima
etal.,2007).Suchfindingsfurtherhighlighttheimportanceofthetrustbuiltbetween
partiesandtheneedforparentstobeabletoaskphysiciansquestionswithoutfearof
judgementordisregard.
Interestingly,afewoftheparticipantsvoicedwantingtobeabletobothsharethe
informationthey’vegatheredconcerningtheirchildren’spolyposisexperiencesandgain
insightintoothers’experiences.Suchaneedseemstobeinlinewiththefactthatmany
oftheparticipants’experiencesfeltinvalidatedbythosewithwhomtheyinitiallyshared
theirstories,includingfamilymembers,friendsandevenphysicians,ultimatelycreating
asenseofisolation.Assuch,itislikelythattheparticipantsareseekinginformation
withinanenvironmentinwhichtheyfeeltheyarelikelytoreceivesupportandashared
understandingoftheirexperiences.Sharedexperiencesasaformofinformationseeking
mayresonatemorewithparentsandmaybemoreeasilyunderstoodthanscholarly
onlinejournalarticlesormedically-focusedInternetpages.AstudybyvanderMarel,
andcolleagues(2009)foundthat57%ofonlinewebsitespostinginformation
concerninginflammatoryboweldiseasescoredeitherfairorpoorusingareadability-
ratinginstrument,indicatingthatthegeneralpopulationmayexperiencedifficulty
understandingmuchoftheinformationontheInternetconcerningtheircondition.
Certainly,theparticipantsdiscussingtheirneedforinteractionwithothersinsimilar
situationsdidnotsuggestthatanecdotalinformationgleanedfromsuchencounters
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
91
couldreplaceconsultationwithmedicalexperts,however,thefindingsindicatethat
information/experiencesharingwithotherindividualsaffectedbypolyposisconditions
offersaformofsupport,care,andrefugefromisolationthatphysiciansalonecannot
offer.Sharingexperiencesincontextssuchasin-personsupportgroupswouldhelp
parentsreducetheirfeelingsofsocialisolation,andself-stigmatization.Itisworthnoting
thataresourceforchildrenandtheirfamiliesseekingtomeetotherfamiliesaffectedby
polyposisconditionsiscurrentlyavailablethroughtheMountSinaiHospital’sZane
CohenCentreforDigestiveDiseaseslocatedinToronto,Ontario(MountSinaiHospital,
2014).Althoughnocurrentresearchexistsontheeffectivenessoftheprogram,my
findingswouldsuggestthattheresourceservesanimportantneedwithinthepolyposis
community.Althoughitisunfortunatethatnoneoftheparticipantsincludedinthestudy
wereawareoftheprogram,Ihavediscussedthepossibilityofbetterinformingpatients
andtheirfamiliesabouttheprogramwiththephysiciansintheGeneticsandPediatric
Gastroenterologyunits.
Participantsdiscussedtheimportantrolegratitudeplaysinframingtheir
experiencesregardingboththeirchildren’shealthandtheirexperienceswiththe
medicalsystem.Withintheliterature,gratitudehasbeenconceptualizedastheemotion
resultingfromrecognizingandappreciatingtheadvantagesorrewardsonehasbeen
givenorexperienced(Wood,Froh,&Geraghty,2010).Withinthecontextofthisstudy,
parentsnotedfeelinggratefulforthemanageablenatureoftheirchildren’scondition
duetoavailablemedicalresourcesandthecomparativelymildnatureofthecondition.
Withinthepositivepsychologyresearch,gratitudehasbeenpositivelylinkedto
well-being.Specifically,McMillen(1999)foundthatindividualsdemonstratinghigh
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
92
levelsofgratitudewhilefacingstressfullifeeventswerelikeliertoutilizecopingskills,
adapttheirbehavioursandreflectontheirstrengthsthanthosewhodidnotexperience
gratitudewhenfacedwithcomparablesituations.Withinhealthyadults,gratitudehas
alsobeennegativelycorrelatedwithanxietyanddepression(Petrocchi&Couyoumdjian,
2016).
Withintheillnessliterature,researchindicatesindividualslivingwithheart
failure,achroniccondition,werefoundtousegratitudeasacopingresource,with
participantsspecificallycitingmedicalresourcesandsocialsupportascausefor
gratitude(Sacco,Park,Suresh,&Bliss,2014).Whilegratitudehasalsobeenfoundtobe
positivelyrelatedtoemotion-focusedcopingandutilizationofpsychologicalresourcesin
familialcaregiversofindividualslivingwithdementia,virtuallynoresearchisavailable
ontherolegratitudeplaysinparentingchildrenwithchronicillness.
Myfindingsrevealgratitudetobeanimportantcopingresourcebywhichparents
wereabletoreframetheiranxietiesanddistress.Althoughnopreviousresearch
explorestheexperiencesofgratitudeofparentsofchildrenwithchronicillness,taken
together,thefindingsofthisstudyseemtobeinlinewiththeavailableprevious
research.
Oneofthemajorthemesrevealedbythedata,Becomingadifferenttypeof
parent,discussestheideaofthe‘ProtectorParent’.Theterm‘ProtectorParent’isusedto
portraytheessenceofthewaysinwhichtheparentsrelatetotheirchildrenasafunction
oftheirconditions.Theparticipantsreportedthattheirchildren’sdiagnosesrepresented
aconcerningthreattotheirhealth,andadoptingthe‘ProtectorParent’identityandrole
wereborneofnecessity.Theroleservesto‘protect’thechildrenfromsocialisolation
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
93
fromsocietyandinvalidationofsymptomsonthepartofphysicians.Myfindingsreveal,
however,thattheroleof‘ProtectorParent’caninfluencetheparents’otheridentities
andchangesocialdynamicswithinthefamilyandsupportsystems.
Asdiscussedintheliteraturereview,participantsreportedplacinghigh
importanceonthevalueofadvocatingtheirchildren’sneedswithinthemedicalsystem
(Dellve,Samuelsson,Tallborn,Fasth,&Hallberg,2006;Zurynski,Frith,Leonard,&
Elliott,2008).Interestingly,thefindingsrevealthatinadvocatingfortheirchildren,
someofourparticipantswereconsciouslymodellingbehaviourstheyhopedtheir
childrenwouldeventuallyadoptforthemselves.Researchindicatesthatapediatric
patient’sdevelopmentalmaturity,whichcomprisescompetenciessuchaspsychological
coping,understandingofillnessself-management,andcommunicationskills,isthemost
predictivefactorinsuccessfultransitionsforinflammatoryboweldiseaseillnessfrom
pediatrictoadultmedicalcare,whichwasmeasuredinquality-of-life,health,and
medicaladherenceoutcomes(Paineetal.,2014).Furthermore,thelevelofinvolvement
byparentsinsupportingtheirchildrenthroughthetransitionwasalsofoundtohave
predictivevalueforsuccessfultransition(Paineetal.,2014).Asdemonstratedthrough
thereportsofourparticipants,transitionbetweenpediatricmedicalcaretoadult
medicalcarecanbeadifficultprocessforbothparentsandchildren.Researchindicates
thatadolescentsoftenlackknowledgeoftheirmedicalhistory,skillsinadvocatingfor
theirneedsandorganizationoftheirmedicalcare(Hait,Barendse,Arnold,Valim,Sands,
Korzenik,,&Fishman,2009;Sebhastian,Jenkins,McCartney,Ahmad,Arnott,Croft,
Russel,&Lindsay,2012).WithintheSocial-EcologicalModelofAdolescentandYoung
AdultReadinesstoTransitiondevelopedbyPaineandcolleagues(2014),parentsserve
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
94
aspartofthechild’smicrosystem,themostimmediatesysteminaffectingthechild’s
experienceandthereforeholdopportunepositionstoinfluencetheirchildren’s
developmentandbehaviour.Furtherresearchsupportstheroleofparentsinteaching
theirchildrentoself-advocate(Daly-Cano,Vaccaro,&Newman,2015),withresearch
alsoindicatingthatover-protectiveparentswhovoicetheirconcernsabouttheir
children’sabilitiestoself-advocateultimatelyhindertheirchildren’slikelihoodof
successindevelopingself-advocatingskills(Dowrick,Anderson,Heyer,&Acosta,2005).
Myfindingssupportthenotionthatparentsplayapivotalroleinpreparingtheir
childrenforsuchatransitionandthattheiradvocacyintheirchildren’searlyyears
benefitstheirchildren’smedicalcarelongaftertheyhavegraduatedfrompediatric
medicalcaresystems.
Thefindingsalsoindicatethatparents’advocacyfortheirchildrenextended
beyondthemedicalsystem.Theparticipantsdiscussedthenecessityofadvocacyintheir
children’sschoolsandplacesofemploymentaswell.Thesefindingsarein-linewith
currentinflammatoryboweldiseaseliterature,whichhasdemonstratedthatteachers
sometimeslackempathytowardsthesymptomsandexperiencesoftheirstudentswith
inflammatoryboweldisease,generallyasaresultoflackofknowledgeandthelackof
visibilityofthecondition(Gordon,2015).Researchalsoindicatesthatparentsof
childrenwithinflammatoryboweldiseasealsoneededtoconsistentlymeetwiththeir
children’steacherstodiscussre-evaluatingtheirchildren’sneedsandaccommodations,
responsibilitiesalsotakenonbysomeoftheparentsinoursamplegroup(Gordon,
2015).
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
95
Theresultsalsosupportpreviousresearchindicatingthatfamiliesofchildren
withchronicillnessesoftenexperienceachangeinfamilydynamics.Specifically,someof
ourparticipantsindicatedtheirchildren’sconditionsexacerbateddifficultiesintheir
marriages,parallelingresultsdemonstratedbyDahlquistandcolleagues(1993).Ina
studyconductedbyEngstrom(1999),familieswithchildrenwithirritablebowel
syndromewerefoundtoexperiencereducedfamilyfunctioning,comparedtofamilies
withchildrenwithdiabetes,familieswithchildrenwithchronicheadaches,andfamilies
withhealthychildren.However,withinthegroupofchildrenaffectedbyinflammatory
boweldisease,theauthorfoundasubgroupofchildrenwhodemonstratedhealthier
psychologicalstatesthanotherswiththesameillness.Thissubgroupreportedhealthier
familyclimates,whereintheyhadaccesstoanindividualintheirfamily,generallytheir
mother,whoencouragedthemtodiscusstheirfeelingsabouttheirconditions(Enstrom,
1999).FindingsbyNicholasandcolleagues(2007,p.6)reconciletheseresults,
demonstratingthroughtheirresearchthatsupportgiventochildrenwithinflammatory
boweldiseasefromtheirparentsismosteffectivewhenit“includesinformationand
emotionalsupportpresentedinanenvironmentofhonest,age-appropriateandsensitive
communication”.Thefindingssupportthisresearch,assomeofourparticipants
indicatedneedingtosupporttheirchildrenincertainwaysinorderfortheirchildrento
acceptsuchsupport.Specifically,twomotherparticipantsofolderchildrenindicated
thattheirchildrenpreferredtoseekhelpfromthembecausetheirmethodsofsupport
weremorevalidatingandlessdirectivethanthatoftheirhusbands.
Priorresearchandthefindingsindicatethatparentsofchildrenwithchronic
illnessesoftentakeonadditionalrolesfortheiraffectedchild,suchasAdvocateand
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
96
Caregiver(Kratz,Uding,Trahms,Villareale,&KIekhefer,2009).Thefindingsrevealthe
importanceofstrongparent-childties,notonlyinbuildinghealthyfamilybutalsoforthe
effectivenessoftheparent’sroleasadvocate.Childrenaffectedbypolyposisconditions
needtofeelcomfortableinconfidingintheirparentswhensymptoms,bothphysicaland
psychological,arise.Emotionalsupportgiventoparentsofchildrenwithchronic
conditionshasbeenconsideredvaluablebyitsrecipients(Linblad,Rasmussen,
Sandman,2005;Liptak,Orlando,Yingling,Theurer-Kaufman,Malay,Tompkins,&Flynn,
2006).
Parentshighlightedtheuncertaintyoftheirchildren’shealthasamajorfactorin
copingwiththenatureoftheirchildren’sconditions.Whilepreviousresearchhas
demonstratedfearofcancertobeasignificantconcernofindividualswithPeutz-Jeghers
Syndrome(Wooetal.,2009),myfindingsrevealthatparentsofchildrenwithpolyposis
conditionsmustcopewithboththeuncertaintyofthedevelopmentofcancer,aswellas
whattheirchildren’sshortandlong-termfutureoverallhealthwilllooklike.As
Participant3discussed,itisthechronicnatureoftheconditionthathasledherto
experiencechronicstress,chronicuncertainty,andchronicworry.
Aspreviouslymentioned,researchindicatesthatindividualswithrarediseases
requireandwantgreatersocialsupportthanthoseaffectedbymorecommonmedical
conditions(Nispen,vanRijken,&Heijmans,2003).Theresultsrevealthattherareillness
community,whilenecessitatinggreaterneedsforsupport,alsoexperiencegreater
challengesandbarrierstoreceivingthesocialsupportthatmeetstheirneeds.Themain
reasonscitedbyparticipantsforthelackofsupportthey’vereceivedincludeddifficulty
connectingwithothersaffectedbythecondition,lackofawarenessandunderstandingof
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
97
theconditioninthecommunity(generalandmedical)aswellasparticipants’own
psychologicalstatesandfearofburdeningothers.
Relevanttothechronicillnessliteratureisthedifferentiationbetween‘invisible’
and‘visible’illnesses,termscoinedbyJoachimandAcorn(2000).Invisibleconditions
arechronicconditionsthatarenotgenerallyexternallyobservableandaretherefore
moredifficultforotherstodetect.Polyposisconditionsfallwithintheinvisibleillness
category,asmostofthesymptomscausedbytheconditionarenotvisiblyapparent.As
such,individualswithinvisibleconditionsareatagreaterriskofhavingtheirsymptoms,
andexperiencesinvalidated,aswasseenintheparticipants’experiencesinadvocating
fortheirchildrenwithinthemedicalcontextandinseekingsupportfromothers.
However,becauseofthe‘invisible’natureoftheinvisibleillnesses,thoseaffectedalso
havetheabilitytochooseforthemselvestowhomtheychoosetodiscloseanddiscuss
thecondition.Inthecaseofpolyposisconditions,choosingtodiscusstheillnessoften
includesdiscussingthe‘disgusting’symptoms,deemedbybothsufferersandsociety,of
boweldiseases(i.e.bloodinstool,diarrhea,constipation)(Hall,Rubin,Dougall,Hungin,
&Neely,2005).Suchunwillingnesstodiscussbodilyfunctionscanmakesocialsupport
seekinguncomfortableforboththoseseekingsupportandthoseprovidingit.
Alsoinlinewithpreviousresearch,myfindingsrevealthattherarenatureof
polyposisconditionsservesasabarrierforreceivingsocialsupport.Thisresearch
illustratesthefactorsimplicatedinthesocialsupportseekingprocessofparentsof
childrenwithpolyposisconditions,supportingthepreviousstudiesindicatingthat
individualsaffectedbyinvisibleillnessesexperiencedifficultyindiscussingthe
conditions(Diener,2001;Stone,2005).
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
98
Thequalityofsocialsupportanditsabilitytomeetrecipients’needsplaysa
pivotalroleinmoderatingitscapacitytoaffectexperiencesofdistress(Sewitchetal.,
2001).Myfindingsrevealthatsocialsupportcanserveasaprotectivefactor,asfoundby
GrootenhuisandLast(1997),withtheabilityofsocialsupporttomeetanindividual’s
needsasparamount.Theparticipantsofthisstudyvoicedaneedtobeabletosharetheir
experienceswithindividualswhohavegonethroughsimilarexperiences,asthe
likelihoodofthoseunfamiliarwiththeconditionsreceivingtheirstorieswithempathy
andunderstandingislow.Itisalsoimportanttonotethatpriorfindingsindicatethatas
anindividual’spsychologicaldistressincreases,thelikelihoodofsatisfactionwiththe
socialsupporttheyreceivediminishes(Hoekstra-Weebers,Jaspers,Kamps,&Klip,
1999).Withinthisstudy,theparticipantsmostvocalabouttheirdissatisfactionwiththe
socialsupporttheyreceivedwerealsothosewhosechildrenweremostsymptomatic.
Thefindingsalsosupportpreviousfindingsinwhichindividualshesitatein
seekingsupportforfearofburdeningothers.Instudyontheexperiencesofchildren
copingwithinflammatoryboweldisease,Nicholasandcolleagues(2007)foundthat
participantsoftenchosenottosharetheirconcernswiththeirfamilymembersinorder
nottotransmittheirworryontoothers.
Itisuncertainwhethergivensocialsupportthatmettheirneeds,theparticipants
indicatingdissatisfactionwiththeircurrentsocialsupportwouldmoreproactivelyseek
tosharetheirexperienceswithothers.Myfindingsfurtherhighlightthepsychological
stateofindividualsneedingsocialsupport,andthebarrierstheyfaceinseekingsuch
resources.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
99
Myfindingsareinlinewithpreviousresearchindicatingthatfearofcancerisa
significantconcernofindividualsaffectedbypolyposisconditions(Wooetal.,2009).
Althoughtheparticipantsinthisstudydiscussedexperiencingdistressing,chronic
concernsregardingthedevelopmentofcancerintheirchildren,suchdistressdidnot
poseasahindrancetoparents’engagementinmedicalmanagement/surveillance
procedures.Suchfindingsarenotable,asthediscoveryofcancerhasbeenfoundtobe
themostreportedreasonforadultsatriskforcolorectalcancernotparticipatingin
screeningprocedures.Myfindingsprovideinsightintohowtheparentsareabletocope
withtheirfearofcancerwhilestillengaginginactivecopingtechniquessuchasensuring
theirchildrenareproperlyfollowedandconsultingwiththeirchildren’sphysicians.The
participantscitedreassurancebytheirphysiciansthatwithappropriatemedical
surveillance,thechancesofcancerdevelopmentlessen.Itwouldseemthatthe
physicians’supportprovidedtheparentswithwaysofcognitivelyreframingtheirfear,
therebyprovidingthemwithhope.
ImplicationsfortheMedicalField
Takentogether,thefindingsfromthisstudycanbeusedbythemedical
communitytobettersupportandservepediatricpatientswithpolyposisconditionsand
theirparents.Whilenotallparentsofchildrenwithpolyposisconditionsmayfindtheir
experiencesreflectedinthestoriescapturedwithinthisstudy,thefindingsthatwere
obtainedcanusedtobothbetterinformthemedicalprofessionandcreateconcrete
objectivesformedicalprofessionals.
Firstly,myfindingssuggestthatinsituationswithpediatricpatients,physicians’
relationshipswiththeirpatients’parentsarejustasimportanttothecareofthechildas
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
100
theiractualrelationshipwiththechild.Inbuildingastrong,collaborativerapportwith
thechild’sparents,thephysicianisdirectlybenefitingthechild’smedicalexperience,
andinmanycases,thechild’shealth.Aspolyposisconditionsaffectthosediagnosedand
theirfamilieschronically,itmaybehelpfulforphysicianstocheckinwithparents
regardingtheirownexperiencesandmentalstateswhileworkingwiththeirpediatric
patients.Asdiscussed,parentsofchildrenwithrareandchronicillnesseshavebeen
showntodemonstrateanelevatedriskfordevelopinganxietyanddepression.Assuch,it
maybehelpfulforphysicianstobeawareofresourcesavailabletoparentsofchildren
withmedicalconditionswhoareexperiencingdifficultycoping.
Continuedattentionshouldbeplacedontherelationshipwiththepatient’s
parents,ensuringthattheirconcernsarevalidatedandaddressed,withspecial
considerationgiventothepossiblydifficultexperiencethatparentsfaceinhavingtheir
children’srareconditiondiagnosed.
Lastly,asmyfindingsrevealthatparentsmakestrongeffortstotraintheir
childrentoself-advocate,wewouldrecommendthatphysiciansremainmindfulofthis
andcollaboratewithbothparentandchildwhendiscussingmedicaltreatments,as
appropriate,especiallyduringthepatient’stransitionbetweenchildandadultmedical
care.
ImplicationsfortheCounsellingDomain
Myfindingssuggestthatcounsellorscouldplayanimportantroleinfacilitating
parents’adjustmenttotheirchildren’sdiagnoses,andsubsequenteverydaylife.Overall,
thisstudyhighlightsimportantthemesthatmayariseinthecounsellingprocess,suchas
fearofthedevelopmentofcancer,changeofidentity,evolvingfamilydynamics,and
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
101
copingstrategies.Importantly,manyoftheparticipantsnotedgratitudeasanimportant
practiceintheireverydaylives,whichhasbeendemonstratedtobeeffectiveinfostering
well-being(Emmons&Stern,2013).Ascopingstyleshavebeenshowntobetransmitted
fromparenttochild,counsellingparentstowardmoreeffectivecopingstylesmay
indirectlyalsobenefitpediatricpatients(Kliewer,Fearnow,&Miller,1996)
Furthermore,asisolationandlackofsupportwerefoundtoberelevantthemesto
theexperiencesoftheparentsinterviewed,ourfindingswouldsuggestthatitwouldbe
bestifthesesupportgroupsareledbyparents.Littleresearchhasbeenconductedon
theuseandefficacyofsupportgroupsforthoseaffectedbygastrointestinaldisorders.
Assuch,thoseseekingtodevelopsupportgroupsforpolyposispatientsandtheir
familiesarelimitedbytheavailableliterature.
Limitations Althoughthisstudyhasrevealedinterestingandimportantfindings,the
limitationsoftheresearchshouldalsobetakenintoconsideration.Firstly,saturationof
themesfromthedatacouldnotbeachievedduetothelimitedsamplesizeofthestudy.It
isworthmentioningonceagain,however,thatsmallsamplesizeisaninherentchallenge
whenstudyingtheexperiencesofindividualswithraredisease(Ettore,2006).
Regardingthetransferabilityofthesefindingstootherparentsofchildrenwith
polyposisconditions,thedemographicsofthisparticularsamplegroupmustalsobe
takenintoaccount.Whilethemajorityofthechildrenconsideredinthisstudywere
diagnosedwithclinicalmanifestationsofeitherJuvenilePolyposisorPeutz-Jeghers
Syndrome,allbutoneparticipant’schildren’sconditionsweredenovoinnature.
Furthermore,noneoftheparentsinterviewedsufferedfromapolyposiscondition
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
102
themselves,whichwouldpossiblyaffecttheirexperiences.Itisalsoimportanttonote
thattheexperiencesofparentswhowereunresponsiveintherecruitmentprocessfor
thisstudymaysignificantlydifferfromthosewhoconsentedtheirparticipationinthe
interviewprocess.Whileitisimpossibletodeterminetheexactreasonsforwhichthe
unresponsiveparentschosenottoparticipateinthestudyatthispoint,theresearchers
questiontheroleavoidanceoftheillnessrealityplayedinthedecision.
Aswithallqualitativeresearch,theprimaryresearcher’interpretativelensmust
beconsidered.Whilesuchafactorisnotnecessarilyalimitationoftheresearch,
variabilityintheinterpretationofthedatamaypresentitselfinfutureresearchbasedon
otherresearchers’ownparticularinterpretationbiases.Futureresearchshouldbe
conductedinordertofurtherelucidatethephenomenarevealedinthisstudyandin
ordertoexplorethedifferencesinexperienceswithparentsofchildreninsimilarbut
differentcircumstances,whereparentsthemselveshavepolyposisdiagnoses,for
example.
AvenuesforFutureResearch
Thisstudyprovidedinsightintotheexperiencesofparentsofchildrendiagnosed
withpolyposisconditions.Continuedresearchinthisdomainwillbekeyindeepening
ourunderstandingoftheseexperiencesanddevelopingmorespecializedmedical
services.Specifically,qualitativeresearchwillaffordresearcherstheopportunitytogain
richdatainascarcelystudieddomain.
Thecurrentstudyshouldbereplicatedusingalargersamplesize,with
participantsrepresentingchildrenwithdenovoandfamilialdiagnoses,andwith
participantsthemselvesdiagnosedwithahereditarypolyposiscondition,inorderto
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
103
betterreflectthediversityofexperienceswithinthepopulation.Furthermore,future
studiesmayconsideraskingeligiblerecruitswhytheyhavechosennottoparticipatein
thestudy,aspossiblereasonsfornon-participation(apathyconcerningtheirchildren’s
condition,strongemotionstiedtotheirchildren’sconditionsordistrustofthemedical
systemoverallduetodifficultexperiences,forexample)warrantreportingandpossible
furtherinvestigationasparentswhochoosenottoparticipateinstudiespossiblydiffer
fromthosewhochooseotherwiseinmeaningfulways.
Thisstudy’sfindingsalsorevealinterestingdirectionsresearchersmaywishto
explore.Manyofthefindingsremaininlinewithpreviousresearchhoweverseveral
significantandnovelresultswerealsodiscovered.Itmaybeofinteresttoresearchers
andcliniciansaliketofurtherstudyroleofgratitudeinillness,high-stresscontexts.The
waysinwhichparentaladvocatestransmittheirskillstotheirchildrenshouldalsobe
furtherexamined,assuchbehavioursplayanimportantroleinthepatientexperience.
Moreover,appliedresearcherscouldexploretheeffectivenessandbestmeansof
implementationofsupportgroupsforindividuals(andtheirfamilies)withrareand
chronicillnesses,asthiswasaneedexpressedbytheparentsofthisstudy.Lastly,as
patientandfamily-centeredcareutilizedbyphysiciansplayedanimportantroleinthe
experiencesofpediatricpatientsandtheirparents,deeperexplorationofthespecific
skillsdemonstratedbyphysicianstovalidatetheexperiencesofindividualsaffectedby
rareandchronicillnessesmayadvanceoverallhealthcarepractice.Whileimportant
practicalapplicationscanbedevelopedfrommyfindings,furtherresearchshouldbe
conductedinordertofurthersubstantiatetheirvalidity.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
104
Conclusions
Parentsofchildrenwithpolyposisconditionsfacemanychallengesincopingwiththeir
children’shealth.Thecurrentstudyexploredtheexperiencesofparentsofchildrenwith
polyposisdiagnoses,gaininginsightintotheirstruggles,needs,appreciation,and
everydaylives.Itishopedthatthisstudyanditsresultswillgivephysicians,researchers
andindividualsunfamiliarwithpolyposisexperiencesanopportunitytopeekintothe
worldsofparentsofchildrenwithchronicandrareconditions.Itistheaimofthisstudy
thatsuchaperspectivewilltranslateintomoreeffectivecareforpediatricpatientsand
theirparents,withmorefocusplacedontheemotionalandpsychologicalneedsof
parents.Furthermore,itishopedthatthemedicalcommunitywillfurtherviewparents
asvaluableresourcesinthecareofchildrenwithpolyposisconditions,withthisstudy
revealingtheparents’uniqueroleinfosteringtheadvocacyskillsoftheirchildren.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
105
References
Annells,1996.Hermeneuticphenomenology:Philosophicalperspectivesandcurrentuse
innursingresearch.JournalofAdvancedNursing,23(4),705-713.doi:
10.1111/j.1365-2648.1996.tb00041.x
Aymé,S.,Kole,A.,&Groft,S.(2008).Empowermentofpatients:lessonsfromtherare
diseasescommunity.TheLancet,371,2048-2051.doi:10.1016/S0140-
6736(08)60875-2
Baskin,C.H.,Forehand,R.,&Saylor,C.(1985).Predictorsofpsychologicaladjustmentin
mothersofchildrenwithcancer.JournalofPsychosocialOncology,3,43-54.doi:
10.1300/J077v03n03_04
Bastani,R.,Gallardo,N.V.,&Maxwell,A.E.(2001).Barrierstocolorectalcancer
screeningamongethnicallydiversehigh-andaverage-riskindividuals.
JournalofPsychosocialOncology,19(3),65-84.doi:10.1300/J077v19n03_06
Bergum,V.(1991).Beingaphenomenologicalresearcher.InJ.Morse(Ed.),Qualitative
nursingresearch(pp.55-71).London:Sage.
Bernstein,K.I.,Promislow,S.,Carr,R.,Rawsthorne,P.,Walker,J.R.,&Bernstein,C.N.
(2011).Informationneedsandpreferencesofrecentlydiagnosedpatientswith
inflammatoryboweldisease.InflammatoryBowelDiseases,17(2),590-598.doi:
10.1002/ibd.21363
Blieker,E.M.A.,Menko,F.H.,Taal,B.G.,Kluijt,I.,Wever,L.D.V.,Gerritsma,M.A.,etal.
(2005).ScreeningBehaviorofIndividualsatHighRiskforColorectalCancer.
Gastroenterology,128,280-287.doi:10.1053/j.gastro.2004.11.002
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
106
Boardman,L.A.,Thibodeau,S.N.,Schaid,D.J.,Lindor,N.M.,McDonnell,S.K.,Bugart,L.J.,et
al.(1998).IncreasedRiskforCancerinPatientswiththePeutz-JeghersSyndrome.
AnnalsofInternalMedicine,128,896-899.doi:10.7326/0003-4819-128-11
199806010-00004
Bonner,S.,Zimmerman,B.J.,Evans,D.,Irigoyen,M.,Resnick,D.,&Mellins,R.B.(2002).An
individualizedinterventiontoimproveasthmamanagementamongurbanLatino
andAfrican-Americanfamilies.JournalofAsthma,39(2),167-179.
Boyatzis,R.(1998).Transformingqualitativeinformation:Thematicanalysisandcode
development.ThousandOaks,California:SagePublications.
Braun,V.,&Clarke,V.(2006).Usingthematicanalysisinpsychology.Qualitative
ResearchinPsychology,3,77-101.doi:10.1191/1478088706qp063oa
Budych,K.,Helms,T.M.,&Schultz,C.(2012).Howdopatientswithrarediseases
experiencethemedicalencounter?Exploringrolebehavioranditsimpacton
patient–physicianinteraction.HealthPolicy,105,154-164.doi:
10.1016/j.healthpol.2012.02.018
Burt,R.W.(2002).Polyposissyndromes.ClinicalPerspectivesinGastroenterology,1(5)
51-59.
Casati,J.,Toner,B.B.,deRooy,E.C.,Drossman,D.A.,&Maunder,R.G.(2000).Concernsof
patientswithinflammatoryboweldisease:Areviewofemergingthemes.
DigestiveDiseasesandSciences,45(1),26-31.doi:10.1023/A:1005492806777
Chevrel,J.,&Gueraud,J.(1975).Troiscasfamiliauxdepolyposejuvenile.Chirurgie,101,
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
107
708-721.
Cima,R.R.,Anderson,K.J.,Larson,D.W.,Dozois,E.J.,Hassan,I.,Sandborn,W.J.,Loftus,E.V.,
&Pemberton,J.H.(2007).Internetusebypatientsinaninflammatorybowel
diseasespecialityclinic.InflammatoryBowelDisease,13(10),1266-1270.Doi:
10.1002/ibd.20198
Chow,E.,Meldrum,C.J.,Crooks,R.,Macrae,F.,Spigelman,A.D.,&Scott,R.J.(2006).An
updatedmutationsspectruminanAustralianseriesofPJSpatientsprovides
furtherevidenceforonlyonegenelocus.ClinicalGenetics,70(5),409-414.Doi:
10.1111/j.1399-0004.2006.00704.x
Chreim,S.,Williams,B.E.,&Hinings,C.R.(2007).Interlevelinfluencesonthe
reconstructionofprofessionalroleidentity.AcademyofManagementJournal,
50(6),1515-1538.doi:10.5465/AMJ.2007.28226248
Clark,N.M.,Gong,M.,Schork,M.A.,Kaciroti,K.,Evans,D.,Roloff,D.,Hurwitz,M.,etal.
(2000).Long-termeffectsofasthmaeducationforphysiciansonpatient
satisfactionanduseofhealthservices.EuropeanRespiratoryJournal,16,15-21.
Coburn,M.C.,Pricolo,V.E.,DeLuca,F.G.,&Bland,K.I.(1995).Malignantpotentialin
intestinaljuvenilepolyposissyndromes.AnnalsofSurgicalOncology,2(5),
386-391.doi:10.1007/BF02306370
Cohen,M.Z.,&Omery,A.(1994).Schoolsofphenomenology.InJ.M.Morse(Ed.),Critical
issuesinqualitativeresearch(pp.136-156).ThousandOaks,CA:Sage.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
108
Colaizzi,P.F.(1978).Psychologicalresearchasthephenomenologistviewsit.InValle,R.
S.,&King,M.(1978).Existential-phenomenologicalalternativesforpsychology.
OxfordUniversityPress.
Connelly,L.M.(2010).Researchroundtable:Whatisphenomenology?MEDSURGNursing,
19(2),127-128.
Creswell,J.W.(2013).Qualitativeinquiry&researchdesign:choosingamongfive
approaches.LosAngeles:SagePublications.
Dahlberg,K.(2006).Theessenceofessences–Thesearchformeaningstructuresin
phenomenologicalanalysisoflifeworldphenomena.Internationaljournalof
qualitativestudiesonhealthandwell-being,1(1),11-19.
doi:10.3402/qhw.v1i1.4904
Dalquist,L.M.,Czyzewski,D.I.,&Jones,C.L.(1996).Parentsofchildrenwithcancer: A
longitudinalstudyofemotionaldistress,copingstyle,andmaritaladjustmenttwo
andtwentymonthsafterdiagnosis.JournalofPediatricPsychology,21(4),541-
554.Doi:10.1093/jpepsy/21.4.541
Dalquist,L.M.,Czyzewski,D.I.,Copeland,K.G.,Jones,C.L.,Taub,E.,&Vaughn,J.K.
(1993).Parentsofchildrennewlydiagnosedwithcancer:Anxiety,coping,and
maritaldistress.JournalofPediatricPsychology,18(3),365-376.doi:
10.1093/jpepsy/18.3.365
Daly-Cano,M.,Vaccaro,A.,&Newman,B.(2015).Collegestudentnarrativesabout
learningandusingself-advocacyskills.JournalofPostsecondaryEducationand
Disability,28(2),213-227.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
109
Dellve,L.,Samuelsson,L.,Tallborn,A.,Fasth,A.,&Hallberg,L.R.(2006).Stressand
well-beingamongparentsofchildrenwithrarediseases:aprospective
interventionstudy.JournalofAdvancedNursing,53(4),392-402.doi:
10.1111/j.1365-2648.2006.03736.x
Diener,J.(2011).Everyonepoopsbutnoonewantstotalkaboutit:Thelived
experiencesofyoungpeoplewithinflammatoryboweldisease.Unpublished
Master'sThesis,Queen'sUniversity.
DiMillo,J.,Samson,A.,Thériault,A.,Lowry,S.,Corsini,L.,Verma,S.,&Tomiak,E.
(2013).Genetictesting:Whenpredictiongeneratesstigmatization.Journalof
HealthPsychology,18(2),125-134.doi:10.1177/1359105313502566
Dockerty,J.D.,Williams,S.M.,McGee,R.,&Skegg,D.C.G.(2000).Impactofchildhood
cancerofthementalhealthofparents.MedicalandPediatricOncology,35,
475-483.doi:10.1002/1096-911X(20001101)35:5<475::AID-MPO6>3.0.CO;2-U
Dowrick,P.W.,Anderson,J.,Heyer,K.,&Acosta,J.(2005).Postsecondaryeducation
acrosstheUSA:Experiencesofadultswithdisabilities.JournalofVocational
Rehabilitation,22(1)41-47.
Drossman,D.A.,Chang,L.,Schneck,S.,Blackman,C.,Norton,W.F.,&Norton,N.J.(2009).A
focusgroupassessmentofpatientperspectivesonirritablebowelsyndromeand
illnessseverity.DigestiveDiseasesandScience,54,1532-1541.doi:
10.1007/s10620-009-0792-6
Dunlop,M.G.(2002).Guidanceongastrointestinalsurveillanceforhereditarynon-
polyposiscolorectalcancer,familialadenomatouspolyposis,juvenilepolyposis,
andPeutz-Jegherssyndrome.Gut,51,21-27.Doi:10.1136/gut.51.suppl_5.v21
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
110
Ellis,C.N.(2004).Polyposissyndromes.InC.N.Ellis(Eds.),InheritedCancerSyndromes
(143-165).NewYork:Springer.
Elwyn,G.,Frosch,D.,Thomson,R.,Joseph-Williams,N.,Lloyd,A.,Kinnersley,P.,Cording,
E.,Tomson,D.,Dodd,C.,Rollnick,S.,Edwards,A.,&Barry,M.(2012).Shared
decisionmaking:Amodelforclinicalpractice.JournalofGeneralInternal
Medicine,27(10),1361-1367.doi:10.1007/s11606-012-2077-6
Emmons,R.A.,&Stern,R.(2013).Gratitudeaspsychotherapeuticintervention.Journalof
ClinicalPsychology,69(8),846-855.doi:10.1002/jclp.22020
Engstrom,I.(1999).Inflmamatoryboweldiseaseinchildrenandadolescents:Mental
HealthandFamilyFunctioning.JournalofPediatricGastroenterologyand
Nutrition,28(4),28-33.
Ettore,E.(2006).ProceedingsfromtheInternationalconferenceoftheNetworkof
PublicHealthInstitutionsonRareDiseases(NEPHIRD).Rome:Rapporti,
ISTISAN.
Finan,M.C.,&Ray,M.K.(1989).Gastronintestinalpolyposissyndromes.
DermatologicClinics,7,419-434.
Giardiello,F.M.,&Trimbath,J.D.(2006).Peutz-Jegherssyndromeandmanagement
recommendations.ClinicalGastroenterologyandHepatology,4,408-415.
doi:10.1016/j.cgh.2005.11.005
Giardiello,F.M.,Welsh,S.B.,Hamilton,S.R.,Offerhaus,G.J.,Gittelsohn,A.M.,Booker,S.V.,et
al.(1987).IncreasedriskofcancerinthePeutz-Jegherssyndrome.NewEngland
JournalofMedicine,316,1511-1514.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
111
Giardiello,F.M.,Brensinger,J.D.,Tersmette,A.C.,Goodman,S.N.,Petersen,G.M.,
Booker,S.V.,etal.(2000).VeryhighriskofcancerinfamilialPeutz-Jeghers
syndrome.Gastroenterology,119,1447-1453.
doi:10.1056/NEJM198706113162404
Glaser,B.G.,&Strauss,A.L.(1967).Thediscoveryofgroundedtheory:Strategiesfor
qualitativeresearch.Chicago,Illinois:AldinePublishingCompany.
Goldenberg,H.,&Goldenberg,I.(2008).Familytherapy:Anoverview.Belmont,California:
ThomsonBrooks/Cole.
Gordon,M.(2015).ParentsasAdvocatesforChildrenwithIBDatSchool.Challenges
SurroundingtheEducationofChildrenwithChronicDiseases.Hershey,
Pennsylvania:IGIGlobal.
Graff,L.A.,Walker,J.R.,&Bernstein,C.N.(2009).Depressionandanxietyininflammatory
boweldisease:Areviewofcomorbidityandmanagement.Inflammatorybowel
diseases,15(7),1105-1118.doi:10.1002/ibd.20873
Grootenhuis,M.A.,&Last,B.F.(1997).Adjustmentandcopingbyparentsofchildrenwith
cancer:areviewoftheliterature.SupportiveCareinCancer,5(6),466-484.doi:
10.1007/s005200050116
Haidle,J.L.,&Howe,J.R.(2016).(Updated[December3,2015])Juvenilepolyposis
syndrome.In:GeneReviewsatGeneTestsMedicalGeneticsInformationResource
(databaseonline).Copyright,UniversityofWashington,Seattle.1993-2016.
Availableathttp://www.genetests.org.AccessedAugust15,2016.
Hait,E.J.,Barendse,R.M.,Arnold,J.H.,Valim,C.,Sands,B.E.,Korzenik,J.R.,&Fishman,L.
N.(2009).Transitionofadolescentswithinflammatoryboweldiseasefrom
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
112
pediatrictoadultcare:asurveyofadultgastroenterologists.JournalofPediatric
GastroenterologyandNutrition,48(1),61-65.doi:
10.1097/MPG.0b013e31816d71d8
Hall,N.J.,Rubin,G.P.,Dougall,A.,Hungin,A.P.S.,&Neely,J.(2005).Thefightfor‘health-
relatednormality’:Aqualitativestudyoftheexperiencesofindividualslivingwith
establishedinflammatoryboweldisease(IBD).JournalofHealthPsychology,
10(3),443-445.doi:10.1177/1359105305051433
Heidegger,M.(1962).Beingandtime(J.Macquarrie&E.Robinson,Trans.).NewYork:
HarperandRow.
Hein,S.F.,&Austin,W.J.(2001).Empiricalandhermeneuticapproachesto
phenomenologicalresearchinpsychology:Acomparison.PsychologicalMethods,
6(1),3-17.doi:10.1037/1082-989X.6.1.3
Hinds,R.,Philp,C.,Hyer,W.,&Fell,J.M.(2004).ComplicationsofchildhoodPeutz-Jeghers
Syndrome:Implicationsforpediatricscreening.JournalofGastroenterologyand
Nutrition,39(2),219-220.doi:10.1097/00005176-200408000-00027
Hoekstra-Weebers,J.E.,Jaspers,J.P.,Kamps,W.A.,&Klip,E.C.(1999).Riskfactorsfor
psychologicalmaladjustmentofparentsofchildrenwithcancer.Journalof
AmericanAcademyofChildAdolescentPsychiatry,38,1526-1535.
doi:10.1097/00004583-199912000-00014
Howe,J.R.,Mitros,F.A.,&Summers,R.W.(1998).Theriskofgastrointestinalcarcinoma
inJuvenilePolyposis.AnnalsofSurgicalOncology,5(8),751-756.doi:
10.1007/BF02303487
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
113
Husserl,E.(1931).Ideas:aGeneralIntroductiontoPurePhenomenology(R.Boyce
Gibson,Trans.).London,England:AllenandUnwinPublishers.
Irvine,E.J.(2004).Patients’fearsandunmetneedsininflammatoryboweldisease.
AlimentaryPharmacology&Therapeutics,20(4),54-49.doi:10.1111/j.1365-
2036.2004.02053.x
Ivanovich,J.L.,&Whelan,A.J.(1997).CancerandPeutz-Jegherssyndrome:Areview.
JournalofGeneticCounselling,6(2),193-206.doi:10.1023/A:1025616203564
Jass,J.A.,Williams,C.B.,Bussey,H.J.R.,&Morson,B.C.(1988).JuvenilePolyposis-A
precancerouscondition.Histopathology,13,619-630.Doi:10.1111/j.1365-
2559.1988.tb02093.x
Joachim,G.&Acorn,S.(2000).Stigmaofvisibleandinvisiblechronicconditions.
JournalofAdvancedNursing,32,243–248.doi:10.1046/j.1365-
2648.2000.01466.x
Johnson,B.H.(1990)Thechangingroleoffamiliesinhealthcare.Children'sHealthCare
19,234–241.
Kazak,A.(1989).Familiesofchronicallyillchildren:Asystemsandsocial-ecological
modelofadaptationandchallenge.JournalofConsultingandClinicalPsychology,
57,25-30.doi:10.1037/0022-006X.57.1.25
Kazak,A.E.,Boeving,C.A.,Alderfer,M.A.,Hwang,W.T.,&Reilly,A.(2005).
Posttraumaticstresssymptomsduringtreatmentinparentsofchildrenwith
cancer.JournalofClinicalOncology,23(30),7405-7410.doi:
10.1200/JCO.2005.09.110
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
114
Kelly,C.S.,MorrowA.L.,Shults,J.,etal.(2000).Outcomesevaluationof
acomprehensiveinterventionprogramforasthmaticchildrenenrolled
inMedicaid.Pediatrics,105,1029–1035.
Koch,T.(1995).Interpretiveapproachesinnursingresearch:TheinfluenceofHusserl
andHeidegger.JournalofAdvancedNursing,21(5),827-836.doi:10.1046/j.1365-
2648.1995.21050827.x
Kratz,L.,Uding,N.,Trahms,C.M.,Villareale,N.,&Kieckhefer,G.M.(2009).Managing
childhoodchronicillness:Parentperspectivesandimplicationsforparent-
providerrelationships.Families,Systems,&Health,27(4),303-313.
doi:10.1037/a0018114
KIiewer,W.,&Lewis,H.(1995).FamilyInfluencesoncopingprocessesinchildrenand
adolescentswithsicklecelldisease.JournalofPediatricPsychology,20(4),511-
525.doi:10.1093/jpepsy/20.4.511
Knight,A.W.,&Senior,T.P.(2006).Thecommonproblemofrarediseaseingeneral
practice.MedicalJournalofAustralia,185(2),82-83.
Kuo,D.Z.,Houtrow,A.J.,Arango,P.,Kuhlthau,K.A.,Simmons,J.M.,&Neff,J.M.(2012).
Family-centeredcare:Currentapplicationsandfuturedirectionsinpediatric
healthcare.MaternalandChildHealthJournal,16(2),297-305.
doi:10.1007/s10995-011-0751-7
LeCam,Y.(2007).Patients’needsandexpectationsconcerningaccesstohealth
services-EURORDIS3rdstudy.ProceedingsfromAccesstoHealthServices:
PatientsNeedsandExpectations.Lisbon,Portugal:EURORDIS.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
115
Lerman,C.,Hughes,C.,Trock,B.J.,Myers,R.E.,Main,D.,Bonney,A.,etal.(1999).
Genetictestinginfamilieswithhereditarynonpolyposiscoloncancer.Journalof
theAmericanMedicalAssociation,282(17),1618-1622.
doi:10.1001/jama.281.17.1618.
Lieberman,D.(1994).Screening/earlydetectionmodelforcolorectalcancer.Cancer,72,
2023-2027.
LincolnYS,GubaEG.Naturalisticinquiry.Vol75:SagePublications,Inc;1985.
Lindblad,B.M.,Rasmussen,B.H.,&Sandman,P.O.(2005).Beinginvigoratedin
parenthood:Parents’experiencesofbeingsupportedbyprofessionalswhen
havingadisabledchild.JournalofPediatricNursing,20,288–297.
doi:10.1016/j.pedn.2005.04.015
Lindor,N.M.,&Greene,M.H.(1998).Theconcisehandbookoffamilycancersyndromes
MayoFamilialCancerProgram.JournalofNationalCancerInstitute,90(14),1039-
1071.doi:10.1093/jnci/90.14.1039
Lindor,N.M.,McMaster,M.L.,Lindor,C.J.,&Greene,M.H.(2008).Concisehandbookof
familialcancersusceptibilitysyndromes.JournaloftheNationalCancerInstitute
Monographs,38,1-93.doi:10.1093/jncimonographs/lgn001
Lindseth,A.,&Norberg,A.(2004).Aphenomenologicalhermeneuticalmethodfor
researchinglivedexperience.ScandinavianJournalofCaringSciences,18,145-
153.doi:10.1111/j.1471-6712.2004.00258.x
Liptak,G.S.,Orlando,M.,Yingling,J.T.,Theurer-Kaufman,K.L.,Malay,D.P.,Tompkins,L.
A.,&Flynn,J.R.(2006).Satisfactionwithprimaryhealthcarereceivedbyfamilies
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
116
ofchildrenwithdevelopmentaldisabilities.JournalofPediatricHealthCare,20,
245–252.doi:10.1016/j.pedhc.2005.12.008
Lopez,K.A.,&Willis,D.G.(2004).Descriptiveversusinterpretivephenomenology:Their
contributionstonursingknowledge.QualitativeHealthResearch,14(5),726-735.
doi:0.1177/1049732304263638
Lynch,H.T.,&delaChapelle,A.(2003).Hereditarycolorectalcancer.NewEngland
JournalofMedicine,348(10),919-932.doi:10.1056/NEJMra012242
MacDonald,D.J.,&Lessick,M.(2000).Hereditarycancersinchildrenandethicaland
psychosocialimplications.JournalofPediatricNursing,15(4),217-225.
doi:10.1053/jpdn.2000.8044
Magni,G.,Messina,C.,DeLeo,D.,Mosconi,A.,&Carli,M.(1983).Psychological
distressinparentsofchildrenwithacutelymphaticleukemia.Acta
PsychiatricaScandinavia,68,297-300.doi:10.1111/j.1600-0447.1983.tb07010.x
Marques,J.F.,&McCall,C.(2005).Theapplicationofinterraterreliabilityasa
solidificationInstrumentinaphenomenologicalstudy.TheQualitative
Report,10(3),439-462.
McCormick,J.B.,Hammer,R.R.,Farrell,R.M.,Geller,G.,James,K.M.,LoftusJr,E.V.,Mercer,
M.B.,Tilburt,J.C.,&Sharp,R.R.(2012).Experiencesofpatientswithchronic
gastrointestinalconditions.HealthandQualityofLifeOutcomes,10(25),1-11.doi:
10.1186/1477-7525-10-25
McMillen,J.C.(1999).Betterforit:howpeoplebenefitfromadversity.SocialWork,(44),
455–468.doi:10.1093/sw/44.5.455
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
117
Merg,A.,&Howe,J.R.(2004).Geneticconditionsassociatedwithintestinaljuvenile
polyps.AmericanJournalofMedicalGenetics,129,44-55.doi:
10.1002/ajmg.c.30020
Merriam,S.B.(1998).Qualitativeresearchandcasestudyapplicationsineducation.San
Francisco:Jossey-Bass.
Merriam,S.B.(2002).Qualitativeresearchinpractice:Examplesfordiscussionand
analysis.SanFrancisco:Jossey-Bass.
Miller,H.,Bauman,L.J.,Friedman,D.R.,&DeCosse,J.J.(1986).Psychosocialadjustmentof
familialpolyposispatientsandparticipationinachemopreventiontrial.
InternationalJournalofPsychiatryinMedicine,16(3),211-230.doi:10.2190/JV6R-
JNEF-49J7-A1FH
MountSinaiHospital,ZaneCohenCentreforDigestiveDiseases.(2014)BuddySystem.
Retrievedfromhttp://www.zanecohencentre.com/gi-cancers/fgicr/kids-
korner/the-buddy-system
Moustakas,C.(1994).Phenomenologicalresearchmethods.ThousandOaks,CA:Sage.
Murray,S.A.,Grant,E.,Grant,A.,&Kendall,M.(2003).Dyingfromcancerindeveloped
anddevelopingcountries:lessonsfromtwoqualitativeinterviewstudiesof
patientsandtheircarers.BritishMedicalJournal,326(7385),368-373.doi:
10.1136/bmj.326.7385.368
Munley,P.H.(1977).Erikson'stheoryofpsychosocialdevelopmentandcareer
development.JournalofVocationalBehavior,10(3),261-269.
doi:10.1016/0001- 8791(77)90062-8
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
118
Nicholas,D.B.,Otley,A.,Smith,C.,Avolio,J.,Munk,M.,&Griffiths,A.M.(2007).Challenges
andstrategiesofchildrenandadolescentswithinflammatoryboweldisease:a
qualitativeexamination.HealthandQualityofLifeOutcomes,5(28),-.Doi:
10.1186/1477-7525-5-28
Nispen,R.M.A.,vanRijken,P.M.,&Heijmans,M.J.W.M.(2003).Levenmeteenzeldzame
chronischeaandoening:ervaringenvanpatiëntenindezorgenhetdagelijks
leven.NIVEL,1-148.
Norberg,A.L.,Lindblad,F.,&Boman,K.K.(2005).Copingstrategiesinparentsof
childrenwithcancer.SocialScience&Medicine,60,965-975.
doi:10.1016/j.socscimed.2004.06.030
Nguyen,G.C.,LaVeist,T.A.,Harris,M.L.,Datta,L.W.,Bayless,T.M.,Brant,S.R.(2009).
Patienttrust-in-physicianandracearepredictorsofadherencetomedical
managementininflammatoryboweldisease.InflammatoryBowelDiseases,15(8),
1233-1239.doi:10.1002/ibd.20883
Packer,M.(1985).Hermeneuticinquiryinthestudyofhumanconduct.American
Psychologist,40,1081-1093.doi:10.1037/0003-066X.40.10.1081
Paine,C.W.,Stollon,N.B.,Lucas,M.S.,Brumley,L.D.,Poole,E.S.,Peyton,T.,...&Mamula,
P.(2014).Barriersandfacilitatorstosuccessfultransitionfrompediatrictoadult
inflammatoryboweldiseasecarefromtheperspectivesofproviders.
InflammatoryBowelDiseases,20(11),2083-2091.
doi:10.1097/MIB.0000000000000136
Parkin,M.D.,Bray,F.,Ferlay,J.,&Pisani,P.(2005).Globalcancerstatistics,2002.AC:
ACancerJournalforClinicians,55,74-108.doi:10.3322/canjclin.55.2.74
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
119
Patel,S.G.,&Ahnen,D.J.(2012).Familialcoloncancersyndromes:Anupdateofa
evolvingfield.CurrentGastroenterologyReports,14,428-438.doi:
10.1007/s11894-012-0280-6
Petrocchi,N.,&Couyoumdjian,A.(2016).Theimpactofgratitudeondepressionand
anxiety:Themediatingroleofcriticizing,attackingandreassuringtheself.Self
andIdentity,15(2),191-205.doi:10.1080/15298868.2015.1095794
Sacco,S.J.,Park,P.L.,Suresh,D.P.,&Bliss,D.(2014).Livingwithheartfailure:
Psychosocialresources,meaning,gratitudeandwell-being.Heart&Lung:The
JournalofAcuteandCriticalCare,43(3),213-218.
doi:10.1016/j.hrtlng.2014.01.012
Samson,A.,Siam,H.,&Lavigne,R.(2007).Psychosocialadaptationtochronic
illness:Descriptionandillustrationonanintegratedtask-basedmodel.
Intervention,127,16-28.
Samson,A.,Tomiak,E.,Dimillo,J.,Lavigne,R.,Miles,S.,Choquette,M.,Chakraborty,P.,&
Jacob,P.(2009).Thelivedexperiencesofhopeamongparentsofachidwith
Duchennemusculardystrophy:Perceivingthehumanbeyondtheillness.Chronic
Illness,5,103-114.doi:10.1177/1742395309104343
Sawyer,M.G.,Antoniu,G.,Toogood,I.,Rice,M.,&Baghurst,P.A.(1993).A
prospectivestudyofthepsychologicaladjustmentofparentsandfamiliesof
childrenwithcancer.JournalofPediatricChildHealth,29,352-356.doi:
10.1111/j.1440-1754.1993.tb00533.x
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
120
Schreibman,I.R.,Baker,M.,Amos,C.,&McGarrity,T.J.(2005).Thehamartomatous
polyposissyndromes:Amolecularreview.TheAmericanJournalof
Gastroenterology,100,476-490.doi:10.1111/j.1572-0241.2005.40237.x
Sebastian,S.,Jenkins,H.,McCartney,S.,Ahmad,T.,Arnott,I.,Croft,N.,...&Lindsay,J.O.
(2012).Therequirementsandbarrierstosuccessfultransitionofadolescents
withinflammatoryboweldisease:differingperceptionsfromasurveyofadult
andpaediatricgastroenterologists.JournalofCrohn'sandColitis,6(8),830-844.
doi:10.1016/j.crohns.2012.01.010
Sewitch,M.J.,Abrahamowicz,M.,Bitton,A.,Daly,D.,Wild,G.E.,Cohen,A.,...&Dobkin,P.
L.(2001).Psychologicaldistress,socialsupport,anddiseaseactivityinpatients
withinflammatoryboweldisease.TheAmericanJournalofGastroenterology,
96(5),1470-1479.doi:10.1111/j.1572-0241.2001.03800.x
Shields,L.,Pratt,J.,&Hunter,J.(2006).Familycentredcare:Areviewofqualitative
studies.JournalofClinicalNursing,15(1),1317-1323.
doi:10.1111/j.1365-2702.2006.01433.x
Shiloh,S.,Koehly,L.,Jenkins,J.,Martin,J.,&Hadley,D.(2008).Monitoringcoping
stylemoderatesemotionalreactionstogenetictestingforhereditary
nonpolyposiscolorectalcancer:Alongitudinalstudy.Psycho-Oncology,17,
746-755.doi:10.1002/pon.1338
Shontz,F.C.(1975).ThePsychologicalAspectsofPhysicalIllnessandDisability.NewYork:
Macmillan.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
121
Sloper.P.(2000).Predictorsofdistressinparentsofchildrenwithcancer:A
prospectivestudy.JournalofPediatricPsychology,25(2),79-91.doi:
10.1093/jpepsy/25.2.79
Solomon,J.L.(1987).FromHegeltoexistentialism.NewYork:OxfordUniversityPress.
Spigelman,A.D.,Murday,V.,&Phillips,K.D.(1989).CancerandPeutz-Jegherssyndrome.
Gut,30,1588-1599.doi:10.1136/gut.30.11.1588
Stone,S.D.(2005).Reactionstoinvisibledisability:Theexperiencesofyoung
womensurvivorsofhemorrhagicstroke.DisabilityandRehabilitation,27(6),293-
304.doi:10.1080/09638280400008990
Sugars,S.(2011).Peutz-Jegherssyndrome:Apatient’sview.FamilialCancer,10,
473-479.doi:10.1007/s10689-011-9453-y
Sullivan,M.J.,Mikail,S.,&Weinshenker,B.(1997).Copingwithadiagnosisof
multiplesclerosis.CanadianJournalofBehaviouralScience/Revuecanadiennedes
sciencesducomportement,29(4),249-256.doi:10.1037/0008-400X.29.4.249
Suls,J.,&Fletcher,B.(1985).Therelativeefficacyofavoidantandnonavoidant
copingstrategies:Ameta-analysis.HealthPsychology,4(3),249-288.doi:
10.1037/0278-6133.4.3.249
Svedlund,M.,Danielson,E.,&Norberg,A.(1994).Women’snarrativesduringtheacute
phaseoftheirmyocardialinfarction.JournalofAdvancedNursing,35(2),197-205.
doi:10.1046/j.1365-2648.2001.01837.x
Syngal,S.,Brand,R.E.,Church,J.M.,Giardiello,F.M.,Hampel,H.L.,&Burt,R.W.2015.ACP
clinicalguideline:Genetictestingandmanagementofhereditarygastrointestinal
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
122
cancersyndromes.AmericanJournalofGastroenterology,110(2),223-263.doi:
10.1038/ajg.2014.435
Tischkowitz,M.,&Rosser,E.(2004).Inheritedcancerinchildren:practical/ethical
problemsandchallenges.EuropeanJournalofCancer,40,2459-2470.
doi:10.1016/j.ejca.2004.06.005
Tomiak,E.M.,Samson,A.,Miles,S.A.,Choquette,M.C.,Chakraborty,P.K.,&Jacob,P.J.
(2007).Gender-specificdifferencesinthepsychosocialadjustmentofparents
ofachildwithduchennemusculardystrophy:Twopointsofviewfora
sharedexperience.QualitativeResearchJournal,7(2),2-21.doi:
10.3316/QRJ0702002
Trivette,C.M.,Dunst,C.J.,Allen,S.,&Wall,L.(1993).Family-centerednessofthe
children’shealthcarejournal.Children’sHealthCare,22(4),241-56.
Utsunomiya,J.,Gocho,H.,Miyanaga,T.,Hamaguchi,E.,Kashimure,A.,Aoki,N.,&
Komatsu,I.(1975).Peutz-Jegherssyndrome:It’snaturalcourseand
management.TheJohnsHopkinsMedicalJournal,136(2),71-82.
vanderEijk,I.,Vlachonikolis,I.G.,Munkholm,P.,Nijman,J.,Bernklev,T.,Politi,P.,Odes,
S.,Tsianos,E.V.,Stockbrügger,R.W.andRussel,M.G.(2004),Theroleofquality
ofcareinhealth-relatedqualityoflifeinpatientswithIBD.InflammatoryBowel
Diseases,10,392-398.doi:10.1097/00054725-200407000-00010
vanderMarel,S.,Duijvestein,M.,Hardwick,J.C.,vandenBrink,G.R.,Veenendaal,R.,
Hommes,D.W.,&Fidder,H.H.(2009).Qualityofweb-basedinformationon
inflammatoryboweldiseases.InflammatoryBowelDiseases,15(12),1891-1896.
doi:10.1002/ibd.20976
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
123
VanHeerden,J.C.(2011).Understandingbeneficiaries’experiencesofqualityin
earlylearningcentres.(Unpublisheddoctoraldissertation).Universityof
Pretoria,SouthAfrica.
VanLier,M.G.F.,Korsse,S.E.,Mathus-Vliegen,E.M.H.,Kuipers,E.J.,vandenOuweland,
A.M.W.,Vanheusden,K.,vanLeerdam,M.,&Wagner,A.(2012).Peutz-Jeghers
syndromeandfamilyplanning:theattitudetowardsprenataldiagnosisandpre-
implantationgeneticdiagnosis.EuropeanJournalofHumanGenetics,20,236-239.
doi:10.1038/ejhg.2011.152
VanLier,M.G.F.,Mathus-Vliegen,E.M.H.,vanLeerdam,M.E.,Kuipers,E.J.,Looman,
C.W.N.,Wagner,A.,&Vanheuden,K.(2010).Qualityoflifeandpsychological
distressinpatientswithPeutz-Jegherssyndrome.ClinicalGenetics,78(3),
219-226.doi:10.1111/j.1399-0004.2010.01469.x
VanLier,M.G.F.,Westerman,A.M.,Wagner,A.,Loorman,C.W.N.,Wilson,J.H.P.,de
Rooij,F.W.M.,…vanLeerman,M.E.(2011).Highcancerriskandincreased
mortalityinpatientswithPeutz-Jegherssyndrome.Gut,60,141-147.
doi:10.1136/gut.2010.223750
Wertz,D.C.,Fanos,J.H.,Reilly,P.R.(1994).Genetictestingforchildrenand
adolescents.JournaloftheAmericanMedicalAssociation,272,875-881.
doi:10.1001/jama.1994.03520110055029
Woo,A.,Sadana,A.,Mauger,D.T.,Baker,M.J.,Berk,T.,&McGarrity,T.J.(2009).
PsychosocialimpactofPeutz-JeghersSyndrome.FamilialCancer,8,59-65.
doi:10.1007/s10689-008-9202-z
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
124
Wood,A.M.,Froh,J.J.,&Geraghty,A.A.(2010).Gratitudeandwell-being:Areviewand
theoreticalintegration.ClinicalPsychologyReview,30,890–905.
doi:10.1016/j.cpr.2010.03.005
Zurynski,Y.,Frith,K.,Leonard,H.,Elliott,E.(2008).Rarechildhooddiseases:how
shouldwerespond?ArchivesofDiseaseinChildhood,93,1071-1074.
doi:10.1136/adc.2007.134940
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
125
AppendixADemographicQuestionnaire
1. Sex:Male___________ Female______________ Other_______________2. Primarylanguageofcommunication:___________________________3. Maritalstatus:4. Pleaseselectyourhighestlevelofeducation:
a. Lessthanhighschoolb. Highschoolc. CEGEP/Technicalcolleged. University(Undergraduatedegree)e. University(Graduatedegree)
5. Doyouyourselfhaveapolyposisdiagnosis?____________________6. Numberofchildren:_______________
a. Numberofchildrenwithpolyposisdiagnosis:_________________i. Pleasespecifythechildren’sages:____________________ii. Pleasespecifyspecificpolyposisdiagnosis:_______________________iii. Pleasespecifyanyotherphysicalormentaldiagnosesyourchildor
havechildrenhavebeengiven:________________________iv. Atwhatagewas/wereyourchild/rendiagnosedwithpolyposis
syndromes?7. Doyouhaveanyrelativeswithpolyposissyndromes?
a. Ifyes,pleasespecifytheirrelationtoyouandtheirdiagnosis. _________________________________________________________________
8. Didyourchildreceivegenetictesting?a. Ifyes,haveyousharedtheresultsofthetestswithrelativesandfriendsof
thechild?9. Howoftendoesyourchildreceivemedicalattention?10. Pleaseindicatethegroupsofindividualsandserviceswhoareapartofyour
supportsystem:a. Partner/spouseb. Familyc. Friendsd. Physicianse. Co-workersf. Psychologicalservicesg. Internetforumsh. Other(Pleasespecify):____________________________________________________
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
126
Questionnairedémographique
1. Sexe:Masculin___________ Féminin______________Autre_______________
2. Languepremièredecommunication:___________________________3. Étatcivil:________________________4. SVPveuillezindiquerleplushautniveaudescolaritécomplété:
a. Primaireb. Secondairec. CEGEP/Collègetechniqued. Université(premiercycle)e. Université(cyclesupérieur)
5. Avez-vouspersonnellementreçuundiagnosticdepolypose?________________6. Nombred’enfants:_______________
a. Nombred’enfantsayantundiagnosticdepolypose:_________________i. Veuillezpréciserl’âgedevosenfants:________________ii. Veuillezindiquerletypedediagnosticdepolypose:-
_______________________iii. Veuillezindiquertoutautrediagnosticdemaladiesphysiquesou
mentalesquevotreenfantouvosenfantsa/ontreçu:______________________________________________________
iv. Àquelâgevotreouvosenfant(s)a-t-il/ont-ilsreçuundiagnosticd’unsyndromedepolypose?________________
7. Est-cequ’unmembredevotrefamillesouffred’unsyndromedepolypose?a. Sioui,quelestsonlienfamilialavecvousetquelestletypedepolypose
dontilsouffre? __________________________________________________________________
8. Est-cequevotreenfantaeuuntestdedépistagegénétique?a. Sioui,avez-vouspartagélesrésultatsdestestsavecdesparentsetdes
amisvotreenfant?______________9. Àquellefréquencevotreenfantreçoit-ildessoinsmédicaux?
__________________________________________________________________10. Veuillezindiquerlesgroupesd'individusetlesservicesquifontpartiedevotre
réseaudesoutien:a. Conjoint(e)/époux(se)b. Famillec. Amisd. Médecinse. Collèguesdetravailf. Servicesdepsychologieg. Forumssurinterneth. Autre(veuillezpréciser):______________________________________
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
127
AppendixBPermissiontoContactParticipantsForm:CoverLetter
DearParent,WearecontactingyouonbehalfoftheChildren’sHospitalofEasternOntarioinourrelativecapacitiesasChiefoftheDepartmentofGeneticsandDivisionHeadofGastroenterology,totellyouaboutastudycurrentlybeingconductedatCHEObyDr.JaniceBarkeyandDr.EvaTomiak’sresearchteam.YouarebeinginvitedtoparticipateinastudytakingplaceintheDepartmentsofGeneticsandPediatricsintheDivisionofPediatricGastroenterology,HepatologyandNutritionatCHEO.Thetitleofthestudyis:TheLivedExperiencesofParentsofChildrenwithaPolyposisDiagnosis.GiventhecareyourchildhasreceivedintheDepartmentofGeneticsand/orPediatricGastroenterology,you,asaparent,havebeenidentifiedasaneligibleparticipantforthisstudy.YourparticipationinthisstudyiscompletelyvoluntaryandtherewillbenoramificationsonthequalityofcareyourchildwillreceiveatCHEO.Thisstudyseekstolearnaboutparents’experiencesoftheirchildren’spolyposisconditions.Yourchild’spersonalhealthinformationwillnotbereleasedintheprocessofthisresearch.Ifyouwouldliketoknowmoreaboutthisstudy,weencourageyoutoreadthestudydescriptiononthefollowingpagesandtocompletethePermissiontoContactParticipantFormfoundonthefollowingpage,oremailAndreaToo,co-investigator,toindicateyourinteresttolearnmoreaboutthestudy([email protected]).Oncetheformiscompletedandmailedbacktousoryouhaveindicatedyourinterestbyemail,aco-investigatorofthestudywillcontactyoutofurtherdescribethestudytoyou.Youwillthenhavetheopportunitytoasktheco-investigatoranyquestionsyoumayhaveaboutparticipationinthestudy.Yourparticipationinthisstudyisvoluntary.Wethankyouforyourtimeandconsideration.Ifyouhaveanyconcernsaboutthisresearchstudy,youcancontacttheChairoftheResearchEthicsBoardat613-737-7600ext.3272.GailGraham,MD,FRCPC,FCCMGChief,DepartmentofGeneticsEasternOntarioRegionalGeneticsPrograms401SmythRoad,Ottawa,ON,K1H8L1Telephone:613-737-7600ext.2620DavidR.Mack,MD,FRCPCProfessorofPediatrics&DivisionHeadofPediatricGastroenterology,Hepatology&NutritionChildren’sHospitalofEasternOntario401SmythRoad,Ottawa,ONK1H8L1Telephone:613-737-7600ext.2516
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
128
Chersparents,Nousvouscontactonsaunomducentrehospitalierpourenfantsdel'estdel'OntariodelapartduchefduDépartementdegénétiqueetduchefdedivisionduDépartementdegastroentérologie.Nousdésironsvousinformerd'uneétudeactuellementmenéeauCHEOparl'équipederechercheduDrJaniceBarkeyetduDrEvaTomiak.VousêtesinvitésàparticiperàuneétudequialieudanslesdépartementsdegénétiqueetdepédiatriedanslaDivisiondegastroentérologiepédiatrique,d'hépatologieetdenutritionduCHEO.Letitredel'étudeest:Lesexpériencesvécuesparlesparentsd'enfantsayantundiagnosticdepolypose.Étantdonnéquevotreenfantareçudessoinsdudépartementdegénétiqueet/oudegastroentérologiepédiatrique,vousavezétéchoisi,entantqueparent,commeparticipantadmissiblepourcetteétude.Votreparticipationàcetteétudeestentièrementvolontaire,etiln'yauraaucuneconséquencesurlaqualitédessoinsquevotreenfantrecevraauCHEO.Cependant,cetteétudechercheàenapprendredavantagesurlesexpériencesdesparentsd’enfantssouffrantdepolypose.Lesrenseignementsmédicauxpersonnelsdevotreenfantneserontpasdévoilésdansleprocessusdecetterecherche.Sivoussouhaitezensavoirplussurcetteétude,nousvousencourageonsàlireladescriptiondel'étudesurlespagessuivantesetdebienvouloircompléterleformulaired’autorisationàcontacterlesparticipantsquevoustrouverezsurlapagesuivante,oudecommuniquerparcourrielavecAndreaToo,co-chercheuse,afind'indiquervotreintérêtetpourensavoirplussurl'étude(atoo086@uottawa.ca.Unefoisquevousaurezremplietquevousnousaurezenvoyéleformulaireouquevousaurezindiquévotreintérêtparcourriel,unco-chercheurdel'étudevouscontacterapourvousdécrirel'étudeplusendétail.Vouspourrezposervosquestionsauco-chercheursurlaparticipationàl'étude.Votreparticipationàcetteétudeestvolontaire.Mercipourvotretempsetvotreattention.Sivousavezdespréoccupationsoudesquestionsausujetdecetteétude,vouspouvezjoindreleprésidentduComitéd'éthiquedelarechercheau:(613)737-7600,poste3272.
_______________________________________GailGraham,MD,FRCPC,chefFCCMG,DépartementdegénétiqueProgrammerégionaldegénétiquedel’Estdel’Ontario401cheminSmyth,Ottawa,ON,K1H8L1Téléphone:613737-7600,poste2620___________________________________________DavidR.Mack,MD,FRCPCProfesseurdepédiatrieetchefdedivisiondegastroentérologiepédiatrique,d'hépatologieetdenutritionCentrehospitalierpourenfantsdel'estdel'Ontario401cheminSmyth,Ottawa,ONK1H8L1Téléphone:613737-7600,poste2516
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
129
AppendixC:PermissiontoContactParticipantsForm:StudyDescription
Title:ThelivedexperiencesofparentsofchildrenwithpolyposisdiagnosesInvestigators:Co-InvestigatorsDr.JaniceBarkey,MD MindyFinkelstein,MSW(PrincipalInvestigator) CathyGilpin,MSc,CCGCDr.EvaTomiak,MDDr.AndréSamsonAndreaToo,BADescription:Aspartoftherecruitmentprocessforastudyexploringtheexperiencesofparentsofchildrenwithpolyposisdiagnoses,wewouldlikeyourpermissiontocontactyoutofurtherexplaintheobjectivesandproceduresofthisstudy.YouarebeinginvitedtoparticipateinthisstudysinceyouhaveachildwhohassoughttreatmentattheChildren’sHospitalofEasternOntarioforapolyposiscondition.Theprimarygoalofthisstudyistobetterunderstandtheexperiencesofparentsofchildrenwithpolyposisdiagnoses.ParentsofchildrenwitheitherclinicalormoleculardiagnosesofPeutz-JeghersSyndromeorJuvenilePolyposisareeligibletoparticipateinthisstudy.Participantsinthisstudywillbeaskedtoparticipateina60-minuteinterviewandtocompleteademographicquestionnaire.Participationinthisstudywillnotaffectyourchild’scurrentorfuturecareattheCHEO.Participationisvoluntaryandyoumaywithdrawfromthestudyatanytimewithnoramifications.Signingthisformonlyindicatesthatyouwouldliketobecontactedinthefuturetolearnmoreaboutthisstudy.Itdoesnotendorseyourparticipationinthestudy.Ifyouagreetobecontactedtolearnmoreaboutthisstudy,weaskthatyoucompleteandsigntheattachedform.Pleasemailtheformbacktotheprincipalinvestigatorusingtheaddressedandstampedenvelope.Ifwereceivetheformwithconsenttobecontactedtohearmoreaboutthisstudy,wewilltelephoneoremailyouwithinformationaboutthestudyandansweranyquestionsthatyoumayhave.Youalsohavetheoptiontoemailoneofthestudy’sco-investigators,AndreaToo,([email protected])tolearnmoreaboutthestudy.Withthisinformation,youcanmakeadecisionaboutyourparticipationinthestudy.Ifwedonothearbackfromyouwithin21days(3weeks),areminderletterwillbemailedtoyoutoseewhetheryouareinterestedinparticipatinginthestudyornot.ThisresearchprojecthasreceivedtheapprovaloftheCHEOResearchEthicsBoard.Ifyouwouldlikemoreinformationabouttheethicalnatureofthisstudyoryourrightsasaparticipantinthisstudy,pleasecontacttheCHEOResearchEthicsBoardat613-737-7600ext.3272.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
130
Anyquestionsregardingthisresearchstudycanbedirectedto:AndreaToo(Co-Investigator)Telephonenumber:514-979-1146Emailaddress:[email protected]
PermissiontoContactParticipantsForm
Title:ThelivedexperiencesofparentsofchildrenwithpolyposisdiagnosesInvestigators:Co-InvestigatorsDr.JaniceBarkey,MD MindyFinkelstein,MSWDrEvaTomiak,MD CathyGilpin,MSc,CCGCDrAndréSamsonAndreaToo,B.A.
▢Iwouldliketobecontactedbytelephone/emailtolearnmoreaboutthisstudy.
▢ IwouldNOTliketobecontactedbytelephonetolearnmoreaboutthisstudy.Name:
Ifyouhaveindicatedyouwouldliketolearnmoreaboutthisstudy,pleaseindicateyourname,contactinformationandthebesttimetoreachyou.
Name: Telephonenumber:Day:() Evening() Emailaddress:
PleaseindicatethemostconvenienttimeforyoutobecontactedbyplacinganX: Monday Tuesday Wednesday Thursday Friday Saturday SundayMorning9:00am-12:00pm
Afternoon12:00pm-4:00pm
Evening4:00pm-8:00pm
SignatureName(PleasePrint)_Date ***PleasereturnthisformbymailingitintheCHEO-addressedstampedenvelopewhencomplete.Thankyouforyourtimeandconsideration.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
131
Formulaired’autorisationàcontacterlesparticipants:Descriptiondel’étudeTitre:Lesexpériencesvécuesparlesparentsd'enfantsayantundiagnosticde
polypose.
Chercheurs:Co-chercheursDrJaniceBarkey,MDMindyFinkelstein,MSW(Chercheuseprincipale)CathyGilpin,MSc,CCGCDrEvaTomiak,MDDrAndréSamson,PhDAndreaToo,BADescription:Danslecadreduprocessusderecrutementpouruneétudeexaminantlesexpériencesdesparentsd'enfantsayantreçuundiagnosticdepolypose,nousaimerionsavoirl'autorisationdevouscontacterpourvousexpliquerplusendétaillesobjectifsetlesmodalitésdecetteétude.Vousêtesinvitéàparticiperàcetteétudepuisquevousavezunenfantatteintdepolyposeayantreçudestraitementsaucentrehospitalierpourenfantsdel'estdel'Ontario.L'objectifprincipaldecetteétudeestdemieuxcomprendrelesexpériencesdesparentsd'enfantsatteintsdepolypose.Lesparentsd'enfantsayantreçusoitundiagnosticcliniqueoumoléculairepourlesyndromedePeutz-Jeghersoudepolyposejuvénilesontadmissiblespourparticiperàcetteétude.Lesparticipantsàcetteétudeserontinvitésàparticiperàuneentrevuede60minutesetàremplirunquestionnairedémographique.Laparticipationàcetteétuden'affecteraenaucuncaslessoinsactuelsoufutursdevotreenfantauCHEO.Votreparticipationestvolontaire,etvouspouvezarrêterdeparticiperàl'étudeàtoutmoment,etce,sansaucuneconséquence.Sivoussignezceformulaire,celaindiqueseulementquevoussouhaitezêtrecontactédanslefuturpourensavoirplussurcetteétude.Ilnecautionnepasvotreparticipationàl'étude.Sivousacceptezd'êtrecontactépourensavoirplussurcetteétude,nousvousdemandonsdebienvouloirrempliretsignerleformulaireci-joint.S'ilvousplaît,envoyezleformulaireauchercheurprincipalenutilisantl'enveloppeadresséeetaffranchie.Sinousrecevonsleformulaireavecvotreconsentementàêtrecontactépourensavoirplussurcetteétude,nousvoustéléphoneronsouvousenverronsdesinformationssurl'étudeetnousrépondronsàvosquestions.Vouspouvezaussienvoyeruncourrielàl'undesco-chercheursdel'étude,AndreaToo,([email protected])pourensavoirplusausujetdel'étude.Aprèsavoireucesinformations,vouspourrezprendreunedécisionéclairéequantàvotreparticipationàl'étude.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
132
Sinousnerecevonspasderéponsedevotrepartd'ici21jours(3semaines),unelettrederappelvousseraenvoyéeafindesavoirsivousêtesintéresséàparticiperàl'étudeounon.Ceprojetderechercheareçul'approbationduComitéd'éthiquederechercheduCHEO.Sivoussouhaitezavoirplusd'informationssurlecaractèreéthiquedecetteétudeouàproposdevosdroitsentantqueparticipantàcetteétude,veuillezs'ilvousplaîtcontacterleComitéd'éthiquedelarechercheduCHEOau613737-7600auposte3272.Toutequestionconcernantcetteétudepeuventêtreadresséesà:AndreaToo(co-chercheuse)Téléphone:514979-1146Courriel:[email protected]
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
133
FORMULAIRED'AUTORISATIONÀCONTACTERLESPARTICIPANTS:Titre:Lesexpériencesvécuesparlesparentsd'enfantsayantun
diagnosticdepolypose.Chercheurs:Co-chercheursDrJaniceBarkey,MDMindyFinkelstein,MSWDrEvaTomiak,MDCathyGilpin,MSc,CCGCAndreaToo,B.A. DrAndréSamson,PhD
▢Jeveuxêtrecontactépartéléphoneoucourrielpourensavoirplussurcetteétude.
▢JeNEveuxPASêtrecontactépartéléphoneoucourrielpourensavoirplussurcetteétude.Nom:
Sivousavezindiquéquevoussouhaitezensavoirplussurcetteétude,s'ilvousplaîtindiquervotrenom,voscoordonnéesetlemeilleurmomentpourvousjoindre.Nom: Téléphone:Jour:() Soir:() Courriel:_________________________________________ Veuillezs'ilvousplaîtindiquerlemeilleurpourvousjoindreeninscrivantunX:
lundi mardi mercredi jeudi vendredi samedi dimanchematin
9h-midi
après-midimidi-16h
soir16h-20h
SignatureNom(encaractèresd'imprimerie)Date ***Veuillezs'ilvousplaîtretournerleformulaireparlaposteauCHEOdansl'enveloppeaffranchie,unefoisleformulairecomplété.Mercipourvotretempsetvotreattention.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
134
AppendixDParticipantInformationandConsentForm
Title:ThelivedexperiencesofparentsofchildrenwithpolyposisdiagnosesYouhavebeeninvitedtoparticipateinaresearchstudybeingconductedbyDr.JaniceBarkeyfromtheCHEODivisionofPediatricGastroenterology,HepatologyandNutrition(DepartmentofPediatrics),Dr.EvaTomiakfromtheCHEODepartmentofGenetics,Dr.AndréSamsonfromtheFacultyofEducationattheUniversityofOttawaandAndreaToo,aMaster’sstudentintheEducationalCounsellingprogramattheUniversityofOttawa.PurposeofthisresearchstudyThepurposeofthisstudyistobetterunderstandtheexperiencesofparentsofchildrenwithpolyposisdiagnoses.Specifically,theresearcherwouldliketobetterunderstandexperiencesofpsychologicaladjustment,navigationofthemedicaldomain,regularillnessmanagementpractices,andexperiencesthatparentsofchildrenwithpolyposissyndromeshavefoundtobeparticularlymeaningfuloncetheirchildwasdiagnosedwithapolyposiscondition.Yourparticipationinthisstudyiscompletelyvoluntary,withnoramifications,eitherbeneficialordetrimental,toyourchild’smedicalcareattheCHEO.Youarefreetowithdrawfromthisstudyatanytime.ProceduresIfyouchoosetoparticipateinthisstudy,oneofthestudy’sinvestigators,Master’sstudentAndreaToo,willmeetyouatCHEOtobegintheinterviewprocess.Duringtheinterviewprocess,theinvestigatorwillaskyouaseriesofquestionsregardingyourexperiencesasaparentofachildwithapolyposissyndrome.Theformatoftheinterviewprocesswillincludeopen-endedquestionssuchas,“Pleasedescribeformethecontextsurroundingyourchild’spolyposisdiagnosis.”Ifthereareanyquestionsyoufeeluncomfortableanswering,youarenotrequiredtodoso.Itisestimatedthattheinterviewprocesswilltake1hourtocomplete.Attheendoftheinterview,youwillbegiventheopportunitytodiscusstheprocesswiththeresearcherandposeanyquestionsyouhave.Youwillalsobegiventheprimaryresearcher’scontactinformationandthatofhersupervisor,shouldyouhaveanyquestionsorcommentsoncetheinterviewhasbeencompleted.Interviewswillbeaudiorecordedandtranscribedfordataanalysespurposes.Transcriptionswillnotincludeyournameoranyotheridentifyinginformation.Transcriptswillbesafelykeptonapassword-protectedcomputertowhichonlytheprimaryresearcherandhersupervisorwillhaveaccess.Priortotheinterviewprocess,youwillbeaskedtocompleteaquestionnaireregardingyourdemographicvariables.Itisexpectedthecompletionofthisformwilltakeapproximately10minutes.Thisformwillaskyoudemographicquestionsaboutyourself,yourfamily,andyourchild’sdiagnosis.Arethereanyriskstoparticipatingintheresearch?
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
135
Therearenosocialorphysicalrisksassociatedwithparticipationinthisstudy.Itispossiblethatsomequestionsintheinterviewmayelicitnegativememories,thoughtsorfeelings;butyouarefreetorefrainfromansweringsuchquestions.Ifneeded,theresearchercanprovideyouwithinformationregardingcontactingourgastroenterologysocialworker,MindyFinkelstein,shouldyouwishtodiscussyourfeelingsfurther.BenefitsYoumayexperienceindirectbenefitsbyparticipatinginthisstudy.Firstly,youmayfindthatsharingyourexperiencestobecatharticandemotionallybeneficial.Secondly,yourparticipationinthisresearchwillhopefullyenabletheprimaryresearchertocontributetotheresearchliteratureonpolyposissyndromes.Thisresearchwillpresentimportantthemesinthelivesofthisstudy’sparticipants,ultimatelyhelpingtobetterpreparemedicaldoctorsandclinicianstoworkwithparentsofchildrenwithpolyposissyndromes.WithdrawingfromthestudyYouarecompletelyfreetowithdrawyourparticipationfromthisstudyatanytime,withnoconsequences.Shouldyoufeelatanypointduringtheresearchprocessthatyourpsychologicalwellbeingiscompromised,youareencouragedtonotifytheresearcherandwithdrawfromthestudy.CompensationParkingorpublictransportationcostsrelatedtoparticipationinthestudywillbecovered.LimitsofConfidentialityAllidentifyinginformationthatyouprovidetotheresearcherwillberemovedfromallfinalreportsrelatedtothestudy.Yournamewillbecodedusingapseudonyminalltranscripts.Allidentifyinginformationwillremainconfidential.Onlyincaseswhereitismandatedbylaworbythecourtswillyourpersonalinformationberevealed.Audiorecordingsandtranscriptswillbesecurelykeptinalockedcabinetintheprimaryresearcher’sofficeforamaximumof7yearstoallowtimeforthedataanalysesandpublicationprocesses.After5years,allfilesrelatingtothisstudywillbedestroyed.YouwillbegivenacopyoftheConsentFormtokeepforyourfiles.Shouldyoubeinterestedinobtainingthefinalresultsofthisstudy,pleasenotifytheprimaryinvestigator.Bysigningthisconsentform,youholdtheresearchinvestigatorstotheirprofessionaldutiesandresponsibilitiesandensurethatyourlegalrightsareupheldasaparticipant.Ifacircumstancearisesinwhichyousufferaninjuryasaresultofyourparticipationinthisstudy,generallegalconventionswillbeapplied.TheCHEOResearchEthicsBoardhasapprovedtheproceduresofthisstudy.TheCHEOResearchEthicsBoardworkstoprotecttherightsofparticipantsinresearchstudies.Ifyouwouldlikemoreinformationconcerningtheethicalstandardsofthisresearch
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
136
process,pleasefeelfreetocontacttheCHEOEthicsReviewBoardat613-737-7600ext.3272.QuestionsQuestionsaboutparticipationinthisstudycanbedirectedtoMaster’sstudentAndreaToobytelephoneat514-979-1146orbyemailatatoo086@uottawa.ca.Theprincipalinvestigatorsofthisstudycanbereachedwiththefollowingcontactinformation:
Dr.JaniceBarkey Dr.EvaTomiak Dr.AndréSamson613-737-7600ext.2516 613-737-7600ext.3512 613-562-5800ext.
4029ConsentIhavereadtheabovestatementsandanyquestionsImayhaveconcerningtheresearchhavebeenanswered.Iamvoluntarilyagreeingtoparticipateinthisstudy.Ihavereceivedacopyofthisthree-pageconsentform.
o Yes,Iagreetobecontactedforfurtherfollow-upquestions.o No,Idonotagreetobecontactedforfurtherfollow-upquestions.
NameofParticipant(Pleaseprint): SignatureofParticipant: Date: Nameofpersonobtainingconsent: Signatureofpersonobtainingconsent: Date:
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
137
InformationsduparticipantetformulairedeconsentementLesexpériencesvécuesparlesparentsd'enfantsayantreçuundiagnosticdepolypose
VousavezétéinvitéàparticiperàuneétudederecherchemenéeparlaDreJaniceBarkeydeladivisiondegastro-entérologiepédiatriqueduCHEO,d’hépatologieetdenutrition(Départementdepédiatrie),laDreEvaTomiakduDépartementdegénétiqueduCHEO,leDrAndréSamsondelaFacultéd'éducationdel'Universitéd'OttawaetAndreaToo,étudianteàlamaîtriseduprogrammeencounsellingéducationneldel'Universitéd'Ottawa.ButdecetteétudederechercheLebutdecetteétudeestdemieuxcomprendrelesexpériencesdesparentsd'enfantsayantreçuundiagnosticdepolypose.Plusprécisément,larecherchechercheàmieuxcomprendrelesexpériencestouchantàl'adaptationpsychologique,lanavigationdansledomainemédical,lespratiquesrégulièresdegestiondelamaladie,etlesexpériencesquelesparentsd'enfantsatteintsdesyndromesdepolyposeonttrouvéparticulièrementsignificativesunefoisqu’ilsontapprisqueleurenfantétaitatteintdecettemaladie.Votreparticipationàcetteétudeestentièrementvolontaire,sansramifications,bénéfiquesounéfastes,àl’égarddessoinsmédicauxquereçoitvotreenfantauCHEO.Vousêteslibredevousretirerdecetteétudeàtoutmoment.ProcéduresSivouschoisissezdeparticiperàcetteétude,l'unedesintervieweusesdel'étude,AndreaToo,étudianteàlamaîtrise,vousrencontreraauCHEOpourcommencerleprocessusd'entrevue.Pendantleprocessusd'entrevue,onvousposeraunesériedequestionsconcernantvosexpériencesentantqueparentd'unenfantayantsyndromedepolypose.Leformatduprocessusd'entrevuecomprendradesquestionsouvertestellesque:«Veuillezs'ilvousplaîtmedécrirelecontexteentourantlediagnosticdepolyposedevotreenfant.»S’ilyadesquestionsaveclesquellesvousnevoussentezpasàl'aise,vousn'êtespasobligéd’yrépondre.Onestimequel’entrevuedureraàpeuprèsuneheure.Àlafindel'entrevue,vousaurezl'occasiondediscuterduprocessusaveclechercheuretdeposertouteslesquestionsquevouspourriezavoir.Onvousdonneraégalementlescoordonnéesduchercheurprincipaletcellesdesonsuperviseur,sivousaviezdesquestionsoudescommentaires,unefoisquel'interviewauraétéréalisée.Lesinterviewsserontenregistréesenformataudioettranscritsàdesfinsd'analysedesdonnées.Lestranscriptionsnecomprendrontpasvotrenomoutouteautreinformationpermettantdevousidentifier.Lestranscriptionsserontconservéesentoutesécuritésurunordinateurprotégéparunmotdepasseauquelseullechercheurprincipaletsonsuperviseuraurontaccès.Avantleprocessusd'entrevue,vousserezinvitéàremplirunquestionnaireconcernantvosrenseignementsdémographiques.Ilestprévuqueceformulaireprendraunedizainedeminutesàremplir.Lesquestionsdeceformulaireporterontsurvous,votrefamilleetlediagnosticdevotreenfant.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
138
Ya-t-ildesrisquesàparticiperàlarecherche?Iln'yapasderisquessociauxouphysiquesliésàvotreparticipationàcetteétude.Ilestpossiblequecertainesquestionsdel'entrevuepuissentfaireressurgirdessouvenirs,despenséesoudessentimentsnégatifs;maisvousêteslibresdenepasrépondreàcesquestions.Sinécessaire,lechercheurpeutvousfournirdesinformationsafindepouvoircontacternotretravailleusesocialeengastro-entérologie,MindyFinkelstein,sivoussouhaitezdiscuterplusendétailsdecequevousressentez.AvantagesVouspourriezretirerdesbénéficesindirectsenparticipantàcetteétude.Toutd'abord,vouspourriezconstaterquelefaitdepartagervosexpériencespeutvousfairedubienetêtrebénéfiqueémotionnellement.Deuxièmement,votreparticipationàcetterecherche,nousl'espérons,permettraauchercheurprincipaldecontribueràlalittératuredanslarecherchesurlessyndromesdepolypose.Cetterecherchepermettradeprésenterdesthèmesimportantsdanslaviedesparticipantsdecetteétude,etenfindecompte,àaideràmieuxpréparerlesmédecinsetlescliniciensdansleurtravailaveclesparentsd'enfantsatteintsdesyndromesdepolypose.Retraitdel'étudeVousêtescomplètementlibredemettrefinàvotreparticipationàcetteétude,àtoutmomentetsansconséquences.Sivousvoussentezàunmoment,pendantleprocessusderecherche,quevotrebien-êtrepsychologiqueestcompromis,vousêtesencouragésàeninformerlechercheuretàmettrefinàvotreparticipationàcetteétude.CompensationLesfraisdestationnementoudetransportencommunliéesàlaparticipationàl'étudeserontcouverts.LimitesdelaconfidentialitéTouteslesinformationsd'identificationquevousfournissezauchercheurserontretiréesdetouslesrapportsfinauxrelatifsàl'étude.Votrenomseracodéenutilisantunpseudonymedanstouteslestranscriptions.Touslesrenseignementspersonnelsresterontconfidentiels.Vosrenseignementspersonnelsneserontrévélésquedanslescasoùceseraitmandatéparlaloiouparlestribunaux.Lesenregistrementsaudioetlestranscriptionsserontconservésentoutesécuritédansunearmoireverrouilléedanslebureauduchercheurprincipalpourunmaximumde7ansafind’avoirletempsd’analyserlesdonnéesetenraisondesprocessusdepublication.Après5ans,touslesdossiersrelatifsàcetteétudeserontdétruits.Vousrecevrezunecopieduformulairedeconsentementquevouspourrezconserverpourvosdossiers.Sivousêtesintéressésàobtenirlesrésultatsdéfinitifsdecetteétude,veuillezs'ilvousplaîtenaviserlechercheurprincipal.
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
139
Ensignantceformulairedeconsentement,voustenezlesenquêteursdelarechercheresponsablesdeleursdevoirsetresponsabilitésprofessionnelles,ainsiqu’àveilleràcequevosdroitssoientrespectésentantqueparticipant.S’ilsurvenaitunesituationdanslaquellevoussouffriezd'uneblessurependantvotreparticipationàcetteétude,lesconventionsjuridiquesgénéralesseraientmisesenapplication.LeComitéd'éthiquederechercheduCHEOaapprouvélesprocéduresdecetteétude.LeComitéd'éthiquederechercheduCHEOtravailleàprotégerlesdroitsdesparticipantsàdesétudesderecherche.Sivoussouhaitezplusd'informationsconcernantlesnormeséthiquedeceprocessusderecherche,vouspouvezcontacterleComitéd'éthiqueduCHEOau613-737-7600poste3272.DesquestionsToutesquestionsausujetdelaparticipationàcetteétudepeuventêtreadresséesàl'étudiantedeMaîtrise,AndreaToo,partéléphoneau514-979-1146,ouparcourrielà[email protected]étudepeuventêtrejointsauxcoordonnéessuivantes:DrJaniceBarkey Dr.EvaTomiak DrAndréSamson613-737-7600poste2516 613-737-7600poste3512 613-562-5800poste4029J’ailulesdéclarationsci-dessusetonabienréponduàtouteslesquestionsquejepourraisavoirconcernantlarecherche.J’acceptevolontairementdeparticiperàcetteétude.J'aireçuunexemplairedeceformulairedeconsentementdetroispages.oOui,j’acceptequ’onmecontactepourd'autresquestionsdesuivi.oNon,jen’acceptepasqu’onmecontactepourd'autresquestionsdesuivi.Nomduparticipant(encaractèresd’imprimerie):_____________________________________Signatureduparticipant:__________________________Date:_________________________Nomdelapersonnequiobtientleconsentement:_____________________________________Signaturedel'obtentionduconsentementdelapersonne:_______________________________Date:_________________________
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
140
AppendixEInterviewProtocol
Tobeverbalizedtoparticipantbyinterviewer.Aspartofastudyontheexperiencesof
parentsofchildrenwithpolyposissyndromes,Iwouldliketoaskyouaboutyour
experiences.Pleasedescribeyourexperienceswithasmuchdetailaspossibleandtothe
bestofyourability.Thereareno‘wrong’answersandnojudgmentwillbeplaced.Ifa
questionmakesyoufeeluncomfortable,pleaseletmeknowandthatquestionwillbe
skipped.Iwouldliketoremindyouthatyouarefreetoquitthestudyatanypointduring
theinterviewwithnorepercussionsfromyourdoctorsortheresearchers.Wouldyou
likeustoclarifyanythingbeforeIbegin?
1. WhenwasyourchilddiagnosedwithPeutz-JeghersSyndrome/JuvenilePolyposis
Syndrome?
2. Pleasedescribeformethediagnosisprocess,thecontextsurroundingit.What
aspectofthediagnosisprocesssticksoutmostinyourmind?
3. Pleasedescribeifandhowyourchild’sdiagnosishasimpactedyourlife.Howhas
theexperienceimpacted:
• Youemotionally?
• Youpsychologically?
• Thewayyouinteractwithfriends,familyand/oryourpartner?
• Thewayyouapproachyourchild’shealth?
• Thewayyouperceiveyourchild’scurrentandfuturehealth?
4. Pleasedescribeyourexperienceinobtainingmedicalcareforyourchild.
• Howdidthemedicalcommunityapproachyourchild’scondition?
• Whatwasyourexperienceinseekinginformationaboutyourchild’s
condition?
• Howdidtheprocessmakeyoufeel?
5. HowhashavingachildwithPeutz-Jegherssyndrome/JuvenilePolyposis
Syndromeaffectedyourfuturefamilyplanningdecisions?
• HashavingachildwithJuvenilePolyposisSyndromes/Peutz-Jeghers
Syndromesaffectedyourdecisiontohavemorechildren?
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
141
6. (Ifapplicable)HowhasyourexperiencewithPeutz-Jegherssyndrome/Juvenile
Polyposissyndromeimpactedthewayyouguideyourchildthroughthe
experience?
• Doyouprovidelessonsfromyourownexperiences?
• Doyouconsciouslymakeanefforttomodelthewaysinwhichyou
hopeyourchildwillcopewiththeillness?
7. Whatadvicewouldyougiveotherparentsofchildrenwithpolyposisexperiences?
8. Wouldyousayyouhaveadaptedtotheillness?Ifyes,how?Ifno,whynot?
• Whathasimpededorfacilitatedthisadaptation?
9. Sinceyourchild’sdiagnosis,whathaschangedforyouasaparent?Asaperson?
Whathasremainedthesame?
10. Whathasbeentheevolutionofyourexperiencefromdiagnosisuntilpresent
time?
• Howhaveyouremotionsandthoughtstowardstheconditionchanged?
• Howhavethechallengessurroundingtheconditionchanged?
11. Whatstrengthsorlessonsdoyoutakeawayfromthisexperienceifany?
12. Whatwouldyouhavelikedmetoask,thatIdidnotask?
Interviewerwillsummarizetheanswersoftheparticipant.
13. HaveIsummarizedyourexperienceswell?
14. Wouldyouliketoaddanything?
15. Wouldyoubewillingtoansweranyfuturequestionsshouldweneedtoclarify
anyaspectsofthisinterview?
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
142
Protocoled'entrevue
Letextedel’entrevuedoitêtretransmisenparolesparl'intervieweur.Danslecadre
d'uneétudesurlesexpériencesdesparentsd'enfantsatteintsdesyndromesde
polypose,j’aimeraisvousinterrogersurvosexpériences.S'ilvousplaîtdécrivezvos
expériencesavecautantdedétailsquepossibleetaumeilleurdevosconnaissances.Il
n'yapasde«mauvaises»réponsesetaucunjugementneseraémisàl’égarddevos
réponses.Sivousn’êtespasàl’aisederépondreàunequestion,veuillezs'ilvousplaîtme
lementionneretcettequestionnevousserapasposée.Jevoudraisvousrappelerque
vousêteslibred’arrêterdeparticiperàl'étudeàtoutmomentaucoursdel'entrevue,et
ce,sansavoiràcraindredesrépercussionsdelapartdevosmédecinsouchercheurs.
Aimeriez-vousnousclarifierquoiquecesoitavantquejecommence?
1.Quandvotreenfanta-t-ilreçulediagnosticdusyndromedePeutz-Jeghers/depolype
juvénile?
2.Veuillezs'ilvousplaîtmedécrirequelaétéleprocessusdudiagnosticetlecontexte
quil'aentouré.Quelaspectduprocessusdediagnosticvousvientleplusàl’esprit?
3.Veuillezmedécrireàquelpointetcommentlediagnosticdevotreenfantaeuune
influencesurvotrevie.Quelaétél’impactdecetteexpérience:
•survousémotionnellement?
•survouspsychologiquement?
•surlafaçondontvousavezinteragiavecvosamis,votrefamilleouvotre
partenaire?
•surlafaçondontvousabordezcequitoucheàlasantédevotreenfant?
•surlafaçondontvouspercevezlasantéactuelleetfuturedevotreenfant?
4.Quelleaétévotreexpérienceencequiconcernel'obtentiondesoinsmédicauxpour
votreenfant.
•Commentlacommunautémédicalea-t-elleabordél'étatdevotreenfant?
•Quelleaétévotreexpérienceencequiconcernelarecherched'informationssur
l'étatdevotreenfant?
•Quelssentimentsavez-vouséprouvédurantceprocessus?
RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents
143
5.Àquelpointlefaitd’avoirunenfantatteintdusyndromedePeutz-Jeghers/depolype
juvénilea-t-ilaffectévosdécisionsfuturesquantàlaplanificationfamiliale?
•Est-cequelefaitd’avoirunenfantatteintdusyndromedePeutz-Jeghers/de
polypejuvénileaaffectévotredécisiond'avoirplusd'enfants?
6.(Lecaséchéant)CommentvotreexpérienceaveclesyndromedesyndromedePeutz-
Jeghers/depolypejuvénileaeuunimpactsurlafaçondontvousguidezvotreenfantà
traverscetteexpérience?
•Retenez-vousdesleçonsdevospropresexpériences?
•Avez-vousconsciemmentfaituneffortpourmodéliserlesfaçonsdontvous
souhaiteriezquevotreenfantfassefaceàlamaladie?
7.Quelsconseilsdonneriez-vousauxparentsd'enfantsatteintsdepolypose?
8.Diriez-vousquevousvousêtesadaptéàlamaladie?Sioui,comment?Sinon,
pourquoi?
•Qu’est-cequiaentravéoufacilitécetteadaptation?
9.Depuislediagnosticdevotreenfant,qu’est-cequiachangépourvousentantque
parent?Entantquepersonne?Qu’est-cequiestrestépareil?
10.Quelleaétél'évolutiondevotreexpérienceenpartantdumomentdudiagnostic
jusqu'àaujourd’hui?
•Àquelpointvosémotionsetvospenséesàl’égarddecetteconditionsont-elles
changé?
•Commentlesdéfisentourantcetteconditionont-ilschangé?
11.Quelssontlespointsfortsoulesleçonsquevousretenezdecetteexpérience,lecas
échéant?
12.Qu'est-cequevousauriezaiméquejevousdemande,etquejenevousaipas
demandé?
L’intervieweurrésumeralesréponsesduparticipant.
13.Ai-jebienrésumévosexpériences?
14.Aimeriez-vousajouterquelquechose?
15.Seriez-vousprêt(e)àrépondreàdesquestionsdanslefutursinousavonsbesoinde
clarifiercertainsaspectsdecetteinterview?