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Oromandibular dystonia after dental treatments: a report of two cases
Soo-Mi Jang1, Yeong-Cheol Cho1, Iel-Yong Sung1, Sun-Young Kim2, Jang-Ho Son1
Departments of 1Oral and Maxillofacial Surgery, 2Neurology, Ulsan University Hospital, College of Medicine, Ulsan University, Ulsan, Korea
Abstract (J Korean Assoc Oral Maxillofac Surg 2012;38:379-83)
Oromandibular dystonia (OMD) is a rare focal form of dystonia caused by prolonged muscles spasms in the mouth, face, and jaw. OMD can develop after dental treatment, as poorly aligned dentures or multiple tooth extraction may cause an impairment of proprioception in the oral cavity, leading to the subsequent development of dystonia. These repetitive involuntary jaw movements may interfere with chewing, swallowing, and speaking. We report here two cases of OMD after dental procedures.
Key words: Tooth extraction, Dystonic disorders, Jaw, Tongue, Focal[paper submitted 2012. 10. 31 / revised 2012. 11. 27 / accepted 2012. 11. 27]
symptomsofthedisorder1,2.ThemechanismofOMDisnot
well-understood.SomecasesofOMDafterdentaltreatment
havebeenreported,althoughthecausalrelationshipbetween
theseproceduresanddystoniaisstillunclear4,5.
Thispaperdescribes2casesofperipherallyinducedOMD.
Sincetheonsetofdystoniaoccurredafteradentalprocedure,
wesoughttodiscusssomeaspectsofclinicalmanifestations,
diagnosticcriteria,mechanisms,andtreatmentoptionsfor
OMD.
II. Cases Report
1. Case 1
A59-year-oldfemalewitha12-monthhistoryofabnormal
jawprotrusivemovementandopeningwasreferredbya
localpractitioner.According toher,herproblemstarted
duringthedentalextractionofherlowerrightmolarteeth
and immediate implantation (Fig.1),and thesymptoms
worsenedovertime.Althoughthelocalpractitionertriedto
adjusttheocclusion,thesymptomwasnotrelieved.Shewas
healthy,withnosignificantmedicalhistoryorfamilyhistory
ofneurologicaldisorder.
Theintra-oralexaminationrevealedseveredentalattrition
ofher residual teethandunstableocclusion.(Fig.2)The
extraoralexaminationconfirmedtheinvoluntaryprotrusive
I. Introduction
Dystonia isaneurologicalmovementdisorderwherein
sustainedmusclecontractionscausetwistingandrepetitive
movementsorabnormalpostures.Thedisordermaybe
hereditaryormaybecausedbyotherfactorssuchasbirth-
relatedorotherphysicaltrauma,infection,poisoning(e.g.,
leadpoisoning),orreactiontopharmaceuticaldrugsparti-
cularlyneuroleptics1.
Oromandibulardystonia(OMD)isfocaldystoniainvolving
themasticatorymuscles,musclesoffacialexpression,and
thoseofthetongueandpharynx.Involuntary,inappropriate,
repetitive,orsustainedmusclecontractionscausevarying
degreesof jawopening,closing,deviation,protrusion,or
retrusionaswellasfacialgrimacing,abnormal tongueor
pharyngealmovement,oranycombinationofthese2,3.
OMDisarare,oftenmisdiagnoseddiseasethatisdifficult
tomanage.Itstreatmenthasbeenlimitedtominimizingthe
Jang-Ho SonDepartment of Oral and Maxillofacial Surgery, Ulsan University Hospital, College of Medicine, Ulsan University, 877, Bangeojinsunhwan-doro, Dong-gu, Ulsan 682-714, KoreaTEL: +82-52-250-8823 FAX: +82-52-250-7236E-mail: [email protected]
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CASE REPORThttp://dx.doi.org/10.5125/jkaoms.2012.38.6.379
pISSN 2234-7550·eISSN 2234-5930
J Korean Assoc Oral Maxillofac Surg 2012;38:379-83
380
Fig. 1. Radiological examination at the initial visit.Soo-Mi Jang et al: Oromandibular dystonia after dental treatments: a report of two cases. J Korean Assoc Oral Maxillofac Surg 2012
Fig. 2. Clinical photo revealing unstable occlusion.Soo-Mi Jang et al: Oromandibular dystonia after dental treatments: a report of two cases. J Korean Assoc Oral Maxillofac Surg 2012
Fig. 3. Electromyography activity was typically reflected as significant high-frequency and high-voltage activity of motor unit potentials with either sus-tained or short-duration bursts of dis-charge patterns at rest when nor mally electrically inactive.Soo-Mi Jang et al: Oromandibular dystonia after dental treatments: a report of two cases. J Korean Assoc Oral Maxillofac Surg 2012
Oromandibular dystonia after dental treatments: a report of two cases
381
OMDwasmade.
2. Case 2
A57-year-oldfemalewithnoprevioushistoryofmove-
mentdisordersandwhowastakingnorelevantmedication
experiencedsuddenonsetofinvoluntaryjawtremor, limi-
tationofmouthopening,andprotrusivetonguemovements
following theextractionofboth lowermolar teethand
immediatepartialdenture6monthsago.(Fig.5)Thesym-
ptomsworsenedovertimeandinterferedwithherspeaking
andeating.
The clinical examination revealed thedenture to be
generallyunstable.Shehadspontaneoustonguemovements
evenwhenshewasnotspeaking.Themovementsworsened
whenherdentureswereremoved.
Shehadnoother relevantmedicaland familyhistory.
Shewas referred to theneurologist for aneurological
exam.Thecranialmagneticresonanceimagingrevealedno
abnormalities,andtheresultoftheneurologicalexamination
wasnormalsaveforthepresenceofabnormalmovement.A
diagnosisoflingualprotrusiveOMDwasmade.
Shewasprescribedbaclofen,analgesics,andocclusal
stabilization appliance.Afteroralmedication andoral
appliancetherapyfor1month,thesymptomimproved,and
shewasreferredtoaprosthodontistfortherepairofherold,
ill-fittingdenture.
Atthefollow-up1monthlater,mildtonguetremorwith
protrusionandjawtremorwereobserved.BTXinjectioninto
thegenioglossusmusclewasrecommended,butthepatient
refusedbecauseoffinancialproblemsandthesymptomsdid
notcauseanydisability.Onlytheoralmedicationofbaclofen
wascontinued.
andopeningmovementwithslightdeviationtowardtheright
side.
Shewasprescribedbaclofen(Baclan;YooyoungPharma-
ceuticalCo.,Ltd.,Seoul,Korea)10mgthreetimesadayand
analgesicsfor thereliefofchronicmusclespasm.Despite
the treatmentwithamusclerelaxantfor threeweeks, the
symptomspersisted.
Shewasreferredtoaneurologistforelectromyography
(EMG).(Fig.3)TheEMGrecordingsrevealedthatherproblem
wasduetoaspasmofthelateralpterygoidmuscle.Botulinum
toxin(BTX)A(Meditoxin;PacificPharma,Seoul,Korea)
wasinjectedatadoseof30unitspermuscleviaanextraoral
approach.(Fig. 4)Aweek later, she showedadefinite
reductionof thedystonicmovement,becomingsymptom-
free.Nosideeffectwasobservedsavefor26mmofactive
rangeofmotion.Adiagnosisofjawopeningandprotrusive
Fig. 5. Radiological examination at the initial visit.Soo-Mi Jang et al: Oromandibular dystonia after dental treatments: a report of two cases. J Korean Assoc Oral Maxillofac Surg 2012
Fig. 4. Botulinum toxin injection was done via the extraoral approach.Soo-Mi Jang et al: Oromandibular dystonia after dental treatments: a report of two cases. J Korean Assoc Oral Maxillofac Surg 2012
J Korean Assoc Oral Maxillofac Surg 2012;38:379-83
382
Thepathophysiologyofdystoniaisunclearbutisthought
tooriginate in the centrallymediateddysregulationof
movementduetodefectinthebasalgangliaparticularlyin
thesensorymotorregionsoftheputamen.Themechanism
ofperipherallyinduceddystonia-asillustratedbyOMD-is
alsobasedonthetheoryofsensorypathwaydisruptionatthe
levelofthebasalganglia15-17.
Dentalinterventioncanberegardedasperipheraliatrogenic
injury.TherehavebeenseveralreportsofOMDfollowing
dentalprocedures3,7,18.Thompsonetal.reportedonewoman
whodevelopedOMDfollowingdentalextraction8.Traumatic
situations in themouth-suchaspoorlyaligneddentures
ormultipleextractions-maycausean impairmentof the
proprioceptionoftheoralcavity,leadingtothesubsequent
developmentofdystonia8,19.Themuscleconditionworsened
over theyear.Note,however, that thecausalrelationship
betweentheseproceduresanddystoniaisstillunclear4.
OMDisdifficult tomanage,andits treatmenthasbeen
limitedtominimizingthesymptomsofthedisorder.Note,
however, that thereareseveral treatmentoptions thatcan
relievesomeof thesymptomsofdystonia,sophysicians
canselectatherapeuticapproachbasedoneachindividual’
s symptoms12,13.Treatment approachesused tomanage
OMDincludemedication,BTX, localanestheticblocks,
dentalappliances,behavioralmodificationandpsychological
support, andsurgicalprocedures12,13.Oralmedication is
theusualfirstlineoftreatment,butthereisnomedication
topreventdystoniaorslowitsprogression.Tetrabenazine,
clonazepam,orotheroraldrugshavebeenassessed in
a systematicway in large studies.The results of oral
medicationforOMDhavebeenlargelydisappointing.Some
authorsfindtheoralmedicationofbaclofentobeusefulfor
OMD10,13,18.
BTXinjectioninto theaffectedmusclewithorwithout
EMGguidanceisasecond-linetherapy7,13.Botoxhasbeen
proventobesuperior tomedical treatmentparticularly in
focaldystonias20.Injectionsofsmallamountsofthischemical
intotheaffectedmusclespreventmusclecontractions,and
they canprovide temporary improvement in abnormal
postures andmovements characterizingdystonia7.The
toxindecreasesmusclespasmsbyblockingthereleaseof
theneurotransmitteracetylcholine,whichnormallycauses
musclestocontract.Theeffectistypicallyseenafewdays
aftertheinjections,anditcanlastforseveralmonthsbefore
theinjectionsneedtoberepeated2,7.
Physical therapy,useofsplints,stressmanagement,and
biofeedbackmayalsohelpindividualswithcertainformsof
III. Discussion
Dystoniaisapersistentposturefromtheco-contraction
ofagonistsandantagonistsand isgenerallyconsidered
partofthespectrumofdyskinesia2,6.Dyskinesiaconsistsof
adverseeffectsincludingdiminishedvoluntarymovements
andpresenceof involuntarymovements,similar toticsor
chorea1.Dystoniaisaneurologicalmovementdisorderrarely
seenbyoralandmaxillofacialsurgeons7whereinsustained
musclecontractionscausetwisting,repetitivemovements,or
abnormalpostures1,2,5.Themovementsmaybepainful,and
someindividualswithdystoniamayhavetremororother
neurologicalfeatures.Severaldifferentformsofdystonia
mayaffectonlyonemuscle,groupsofmuscles,ormuscles
throughoutthebody1,8,9.Thecauseofmajorityofthecases
isnotknown.Hereditaryorother factors suchasbirth-
related,otherphysicaltrauma,infection,poisoning(e.g.,lead
poisoning),andreactiontopharmaceuticaldrugsparticularly
neurolepticsarepresumedtobethepossiblecauses8-10.
Therearedifferentwaysofdystoniaclassification-itcan
beclassifiedbyetiologyintoprimary(alsoreferredtoas
idiopathic,inherited,orfamilial)andsecondaryforms(also
referredtoasacquired).Primarydystoniareferstodystonia
thatdoesnothaveaclearcause.Manyinstancesofdystonia
are idiopathic.Thesecondaryformofdystoniadevelops
duetoenvironmentalfactors, long-termneurolepticdrugs
(tardivedystonia),neurologicaldisease,braininjury,Wilson’
sdisease,andperipheraltrauma.Themostcommonformis
tardivedystonia,whichdevelopsasasideeffectoflong-term
treatmentwithantipsychoticdrugs11.
Dystoniacanalsobeclassifiedbyageofonset,anatomically
byregionofdistributionsuchasfocal,segmental,multifocal,
andgeneralized,andfurtherintotheaffectedbodyparts2,12,13.
OMDisfocaldystoniainvolvingthemasticatorymuscles,
musclesoffacialexpression,andthoseof thetongueand
pharynx.Involuntary,inappropriate,repetitive,orsustained
musclecontractionscausevaryingdegreesofjawopening,
closing,deviation,protrusion,orretrusionaswellasfacial
grimacing,abnormaltongue,orpharyngealmovementorany
combinationofthese,withthemuscleconditionworsening
over theyear2,3.OMDisclassifiedas jawopening, jaw
closing,jawdeviating,lingualdystonia,orcombinationsof
these2,4,8.
FocalOMDisextremelyrare.TheprevalenceofOMD
varies,reportedlyashighas6.9casesper100,000.Women
seemtobeaffectedmorefrequentlythanmen,withtheonset
typicallybetweentheageof45and70years14.
Oromandibular dystonia after dental treatments: a report of two cases
383
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dystonia12,13.Surgicaltherapiesincludeperipheralandcentral
surgery.Peripheraldenervationormyectomy is seldom
neededsinceOMDusuallyrespondswelltoBTXandmust
bedelayedwhileothertreatmentoptionsareeffective12,13.
These2peripherallyinducedOMDcasesfolloweddental
procedure,withnofamilyhistoryofmovementdisorders,
organicbrainlesion,orexposuretoneurolepticdrugs.The
onlypredisposingfactorwastherecentdentalextractionand
prosthetic treatment.These2patientsexperiencedashort
latencyperiodbetweentheextractionandonsetofdystonia,
andthesymptomsworsenedovertime.Thecloseassociation
oftime,locationoftheprocedure,andonsetofsymptoms
suggeststhattheonsetofdystoniamayhavebeencausedby
thedentalintervention.
Becauseof its rareoccurrence,patientswithOMDare
probablyoftenmisdiagnosed,or theirdiagnosismaybe
delayed.Consequently, thesepatientsmayalso receive
incorrectdental treatment,andthesymptomsmayworsen
overtheyears.
OMDmaybe caused or aggravated by someof the
otherdentalandprostheticproblems.Thus, inplanning
thedental treatmentandprocedure, thesubtypeofOMD
andthepossibledystonicmovementsandforcesshouldbe
considered.
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