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1 5 0 2003 BJU INTERNATIONAL | 91, 150154 | doi:10.1046/j .1464-4096.2003.04001.x
ASSESSING QUALITY OF LIFE IN CHILDREN and ADOLESCENTS
E.W. GERHARZ
et al.
Current approaches to assessing the quality of life inchildren and adolescentsE.W. GERHARZ, C. EISER* and C.R.J. WOODHOUSEDepartment of Urology, Julius Maximilians University Medical School, Wrzburg, Germany, *Department of Psychology, University of Sheffield, and
The Institute of Urology & Nephrology, Royal Free and University College Medical School, London, UK
SUMMARY
Urologists are well aware of the importance
of the quality of life (QoL) in determining thesuccess of their treatments. The public are
increasingly aware of this aspect of medicine.The advent of fetal ultrasonography has madeknowledge of QoL in long-term survivors of
congenital anomalies essential in counsellingabout pregnancy termination. It is becoming
mandatory that clinical trials involving adultsinclude an assessment of QoL. For children,measuring QoL has been restricted to life-
threatening conditions such as cancer ortransplantation medicine. Measuring QoL isdifficult in children and adolescents, and this
is reflected in the few suitable instrumentsavailable. The development of sexualitycontributes a further difficulty as many
anxieties typical of those with genitalanomalies may be common to all teenagers.
Several instruments rely on the opinions of aproxy (parent or carer) but self-assessment bythe child is preferable where possible. The
need for child self-assessment is supported byfinding little concordance between child and
proxy assessments. While measuring QoL ischallenging, we provide a substantial case forgreater consideration of QoL in paediatric
urology.
INTRODUCTION
In these times of disappearing medicalpaternalism, in which increasingly self-confident patients search the Internet before
consulting their doctor, it seems almostredundant to state that decision-making inmodern medicine must include consideration
of the patient's perspective. At the same time,the traditional sanctity of life ideology
governing medicine is under attack. The resultis a growing acceptance that doctors mustnot only keep people alive, but also pay
attention to the quality of life (QoL) ofsurvivors [1]. There is a consensus thattraditional endpoints such as the reduction in
symptoms and improved survival areinsufficient for evaluating medical outcomes.
The inclusion of more holistic patient-
orientated variables is necessary.
As a reflection of this the concept ofmultidimensional health-related QoL isincreasingly important, arguably becoming
the most widely researched subject in thesocial and clinical sciences. However, what
seems to be self-evident in the adult world isa rather recent development in children [2].Although QoL is important for children with
chronic diseases or congenital disorders,interest in children's QoL did not gain
momentum until the 1980s [3].
The increasing attention given to measuring
children's QoL can be attributed primarily tothe changing epidemiology of childhooddisease, from acute to chronic and from
incurable to curable or palliative. Where itis possible to manage but not cure a disease,it is important to determine how far
treatment and disease compromise the child'sQoL. In this way, informed judgements can be
made about whether or not treatment isappropriate, and critically, where there is achoice, which might be the best option for the
child [4].
Measures of QoL are used in routine audit
work or evaluating interventions, andcertainly need to be considered when
making end of life decisions, i.e. when it isappropriate to withhold treatment becausethe anticipated QoL is so poor. They may assist
decisions about rationing of resources. Theadvent of fetal ultrasonography hasfacilitated the identification of a range of
major but not life-threatening anomaliesfor which parents require counselling
before deciding whether or not toterminate the pregnancy. At other times itmay simply be useful to understand the
child's perception of illness and its effect ondaily life.
These considerations apply to a broad rangeof issues in paediatric urology. Whereas the
success of the numerous reconstructiveprocedures for urogenital abnormalities hasbeen detailed meticulously in terms of
mortality and physical morbidity, littleresearch has explored the psychologicalimpact and QoL associated with surgical
treatment.
WHY QOL MUST BE MEASURED
Children with chronic illness have a higher
incidence of psychiatric, psychosocial,educational, and emotional problems than
have healthy children. The effects are variablenot only with the visibility or invisibility of thecondition, but with a whole host of additional
external and internal factors [5,6].
Explanations for these findings have centredon generic variables which affect childrenregardless of the specific nature of their
disease. These include school absence, whichpotentially compromises school achievementand social relationships; reduced
opportunities to take part in sports and socialactivities; and changes in family relationships,especially where parents are responsible for
home-based care. In addition to these genericfactors, QoL is compromised depending on
the characteristics of the specific condition.Thus, in diabetes, the need for regular bloodtests, insulin injections and vigilance about
diet are considered major obstacles toachieving a good QoL. In cystic fibrosis, diet is
again important, as is daily physiotherapy.
In the context of urological disorders there is
a complex interplay between QoL and thecontrol of evacuation. Children withurological conditions may be house-bound, as
nurseries may not accept incontinentchildren. They may be unable to play games
because of urinary leaks. Physically disabledchildren may not be able to take part in sports,except in similarly disabled groups. Hidden
problems such as incontinence may have amore damaging effect than overt ones;incontinence may be a bar to swimming
because of the fear of contamination. Thisforces a child to admit to a problem that
might otherwise have remained private. Fecalincontinence is a particular difficulty becauseof the smell. A child who needs special
permission to leave class because of urinaryurgency is singled out as different.
The expression of QoL also depends on thechild's age. In the passage to adult life,
children must take control of their lives. In the
chronically sick child, a very difficult moment
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comes when the child wishes to makedecisions that previously have been made by
the parents. As a part of this process theindividual must be made aware of the natureof the disease.
As sexuality becomes more important duringadolescence, exstrophy patients and others
with genital anomalies may experienceparticular challenges to achieving a
completely fulfilled life [79]. Very badlydeformed adolescents might wish that theyhad never been born. A pregnant mother
might opt for termination of pregnancy if thefetus had spina bifida, but the baby may be asloved as any other if she did elect to continue
with the pregnancy. Only 8% of youngwomen with spina bifida would accept a
termination if they were pregnant with a
similarly affected fetus [10]. Scientific andanecdotal evidence shows that selective
termination, based on a perception of long-term QoL, is reducing the incidence of babieswith spina bifida, exstrophy and prune-belly
syndrome [11]. Even in cases of PUV there is atrend towards termination of pregnancy, even
though there is no formal evidence thatthe QoL of patients with this condition isimpaired. In contrast, there are many
examples of handicapped adolescents leadingheroic lives.
A particular difficulty arises if the adolescent
makes a decision that the parents perceive tobe wrong. In rare cases where treatment,especially surgery, is needed, a real conflictcan develop. Occasional cases have been
referred for judicial review. However, morecommonly the problem concerns a QoL issue,such as the need to become continent or
develop sexuality. Here an objective QoLmeasure is of no help; if the adolescent does
not perceive the need for treatment, there isno point in pursuing the matter. Conversely, itis wrong for parents to pressurize their
offspring against treatment, especially in
relation to sexual therapy. Adolescents withspina bifida are sometimes over-protectedby their families and such practice reducesthe likelihood of independence. However,
even with a cosseted upbringing, activeprogrammes to teach independence in earlyadult life can be successful and can improve
QoL [12,13].
Groups of adolescents have little tolerance fornonconformists. A year in the life of anadolescent is a long time and it is a common
observation that lives can be devastated andtriumphant in a short space of time, often
as a result of changes in peer pressure and
behaviour. The obvious consequence is thatcomparison with objective criteria is almostimpossible.
In adulthood every effort must be made to
establish independence from parents andclose family. Education must aim at financial,physical and emotional freedom, if at all
possible. By the end of adolescence theremust be a plan for adult life that takes intoaccount the needs of the patient and of the
family.
There is one area in which adolescents andadults have the same desire, i.e. the freedomfrom lavatories, by having complete
continence. Incontinence of urine, feces andflatus are severe burdens, easily measurable,which limit the QoL in all ages. In adolescents
with anorectal agenesis, poor continencecorrelates strongly with mental health
problems and psychosocial disorders [14].
THE CHALLENGE OF MEASUREMENT
Assessing QoL in children poses uniqueproblems [4]; children do not share adultviews about the cause and treatment of
illness. They may interpret questionsdifferently, and adopt a different timeperspective about the course of a disease. In
addition, their abilities to use rating scales,
understand the language, and generallycomplete lengthy questionnaires of the typeused in adult work, may be compromisedby age and cognitive development.
Understanding QoL in children will thereforenot be achieved by scaling down ideasdeveloped through work with adults. To
match the features unique to childhood andadolescence a joint WHO/International
Association for Child Psychology andPsychiatry Working Party [15] proposed in1993 that new measures should be child-
centred, use subjective self-reporting where
possible, be age-related or at leastdevelopmentally appropriate, have a generic
core and specific modules, and emphasisehealth-enhancing aspects of QoL. The
measures should also be cross-culturallycomparable and include the child's concept ofhealth and illness (which may depend on
developmental stage and culturalbackground).
As children are often regarded as unreliablerespondents, early attempts to rate children's
QoL were based on mothers' reports. However,children and parents do not necessarily share
similar views about the impact of illness.
Obtaining data directly from the children maytherefore be more desirable. However, thereare circumstances where children are too
young or ill to answer for themselves. Allmeasures should therefore include provision
for proxy ratings. In these situations, a criticalquestion is how far ratings between child andproxy are similar. In practice, children and
parents do not necessarily share the sameperspective or experience of an event. Indeed,children may not want to share all their
thoughts and feelings with their parents.Furthermore, parents have their own
concerns which may colour their perceptionof the child's QoL.
Ideally, all measures should include versionsfor self-completion by children as well asparallel forms for proxies. Questions should be
re-phrased away from the search forconcordance and towards understanding
the circumstances in which parents andchildren agree (or disagree) about thechild's QoL.
THE VALUE OF ADULT QOL MEASURES IN
CHILDREN
The issue of QoL has received considerableattention in adult work. Given this, it istempting to take an adult measure, change
the wording a little, and then use it for work
with children. There are several problems withthis approach. Adult measures tend to be too
long for children; rating scales may be toocomplex, requiring sensitivity judgements
beyond the child's abilities. In addition, itcannot be assumed that the domains of QoLconsidered appropriate for adults are
necessarily the same for children. Central tochildren's QoL are issues about the ability tofulfil age-related activities. For adolescents,
any comprehensive assessment of QoL mustinclude issues of body image, autonomy and
aspirations for the future. These issues are
rarely included in adult measures. For youngerchildren, issues about attachment to the
family, relationships with peers and siblingsand developing cognitive competence arelikely to be important.
ASSESSMENT OF QOL IN CHILDREN AND
ADOLESCENTS: THE CURRENT STATUS
An update of a comprehensive literaturesearch [16] based on work between 1980 and
1998, using key electronic databases,illustrates that much needs to be done to
improve QoL assessment in children. While
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over 30 000 publications relevant to QoL inmedicine were identified, only 12% were
related to children, mostly involvingtheoretical and conceptual work. Over 90% ofempirical studies used parents or clinic staff
as reporters of the child's QoL. The conditionsmost frequently studied (oncology andtransplantation medicine) tended to be those
with significant mortality rates and wheretreatment requires a high cost and levels of
care. Other chronic conditions that receivedattention included asthma, epilepsy, diabetesand rheumatism. QoL was more often
assessed for older children (1318 years) thanfor those aged 612 years. Many studiescovered a wide range of ages with no
stratification of the results. Interviews,questionnaires, clinical and social indicators
have been used to assess QoL.
A recent report commissioned by the NHS
Health Technology Assessment Programme[4] was conducted to determine the scope andquality of measures currently available for
work with sick children. A broad searchstrategy was devised to provide a
comprehensive list of publications that haveused direct or proxy measures for QoL forchildren and adolescents. Of 137 articles
identified, 43 involved the development of anew measure, 79 reported furtherdevelopment and application of measures,
and 15 assessed QoL on the basis of a battery
approach, i.e. using several measures. Of the43 measures retrieved, 19 were generic and24 were disease-specific. Only three genericmeasures [1719] and two disease-specific
measures [20,21] satisfied basic psychometriccriteria. Many measures were identified forasthma (four), cancer (five) and epilepsy
(four). Sixteen measures allowed forcompletion by children and parent or
caregiver.
Measures were developed in the USA (18), the
UK (eight), Canada (eight) and the
Netherlands (two). Single measures weredeveloped in Germany, Israel, Spain, Sweden,Norway, and Finland. The measures weredescribed as QoL (30), health status (eight),
functional status (two), perception of illness,life satisfaction, and quality of well-being(one each).
Among the generic measures, one was
targeted at children aged 05 years, six atchildren across a broad age range, two atchildren in middle childhood ( 611 years),
four at adolescents, and four at children from8 years to late adolescence; in addition, two
measures were based on adult measures.
DISCUSSION
While it is important to recognize the
methodological difficulties inherent inmeasuring QoL, over-emphasis on these
problems must not be used as an excuse tofocus on hard or clinical outcomes. In thefinal analysis, the goals must be to improve
quality as well as quantity of life.
For those considering measuring QoL in their
clinical or research work, decisions about how,
whatand whereneed to be made. Generic
measures are assumed to be preferable whendecisions need to be made about allocatingresources from public health perspectives. In
contrast, disease-specific measures havemerit when assessing the impact of a changein treatment, or when evaluating outcomes in
clinical trials. Among disease-specificmeasures, asthma, cancer and epilepsy have
received most attention. For children withmany other conditions it is only possible torate QoL using a generic measure.
Disease-specific measures are inappropriate
where a child has more than one condition.Furthermore, the low incidence of someconditions (e.g. intersex, bladder exstrophy)
may preclude the development of disease-specific measures. Wallander et al. [22] arguethat disease-specific measures are little more
than indictors of how the individual perceives
the illness.
There is therefore a need to understand therelation between generic and disease-specific
QoL. The development of a core genericinstrument supplemented by disease-specificmodules may be the solution. This allows for
direct comparison between illness samples,and additional information to be obtained
about specific disease.
It is also imperative to accept that both child
and proxy ratings have value. The question is
to clarify how differences in the perception ofQoL arise between the child and proxy. This
also applies to ratings of QoL made byteachers and other proxies. Parents may be
influenced by other children they know, theirexpectations and hopes for the child,additional life stresses, and their own mental
health.
Based on findings that children and parents
differ in their understanding of illness andtreatment, there is a widely endorsed view
that children should rate their own QoLwherever possible. Despite this, many
measures rely exclusively on parental
reporting. A few measures provide parallelforms for completion by both child andparent. These may be the measures of choice
where children are well and able to rate theirQoL.
Measures are typically targeted at childrenacross a broad age range, with very few
measures available for those aged
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and satisfactory option, but in fact requiresextensive work to establish true comparability,
given cultural differences in the meaning ofillness, relationship between parents andchildren, and organization of healthcare
services. Lack of attention to cross-culturalissues must be addressed before blindlyadopting norms based on other cultural,
social or illness groups.
Thus, we conclude that there aremethodological limitations in all themeasures currently available. However, we
feel strongly that measuring QoL should notbe abandoned. The refinement of existingmeasures and the future development of
more sophisticated measures dependscrucially on experience gained in using those
that are currently available. This interface
between constructing measures and clinicalapplication requires multidisciplinary
collaboration; the potential for mutual benefitis enormous.
An improved understanding of thepsychosocial and QoL aspects of urogenital
abnormalities and their treatment mayprovide a scientific basis for aninterdisciplinary concept in the long-term
care of such patients. In 1999, Schober [26]stated that no published study has adequatelyaddressed the QoL of intersexuals. In a
systematic overview of outcome studies in
patients with bladder exstrophy, only 10 of1260 articles (0.8%) focused on the mental orpsychosocial aspects, and most of these hadserious methodological deficiencies [7]. Many
studies state that outcomes wereunsatisfactory without stating howsatisfaction was measured.
There is no doubt that much needs to be done
to improve the quality of QoL measures, andhence the status of this work in clinicalpractice and research. However, recognition
of QoL has done much to raise the profile of
children's views about the treatment andorganization of care. A recognition of theshortcomings of currently available measuresmust not be used as a reason to ignore QoL
issues. At the least, attention to QoL hasemphasized the need to consider theoutcomes of paediatric medicine in terms of
the whole child rather than a narrow range ofclinical indicators.
CONCLUSIONS
All currently available measures of QoL have
limitations, but it is essential that attempts be
made to use QoL measures in clinical research,to gain experience that will guide thedevelopment of a second generation of more
sophisticated instruments. QoL researchersand clinicians need to cooperate to face this
exciting challenge.
ACKNOWLEDGEMENTS
Christine Eiser is supported by Cancer
Research-UK. The review of QoL measures forchildren was commissioned by the NHS
Health Technology Assessment (HTA)Programme. Elmar Gerharz was supported bythe Incontinence Research Trust. The views
and opinions expressed in this paper reflectthose of the authors and do not necessarily
reflect those of the HTA.
REFERENCES
1 Browne JP. Health-related quality-of-life
studies in urology. conceptual andmethodological considerations. World JUrol 1999; 17: 1938
2 Gerharz EW. Quality of life research inchildren: Fashion or future? Dial Ped Urol
1997; 20: 123 Eiser C, Morse R. The measurement of
quality of life in children: past and future
perspectives. Dev Behav Ped2001; 22:
248564 Eiser C, Morse R. Quality-of-life measures
in chronic diseases of childhood. Health
Technol Assess2001; 5: 1147
5 Beck AL, Nethercut GE, Crittenden MR,Hewins J. Visibility of handicap, self-concept and social maturity among
young adult survivors of end stage renaldisease. J Dev Behav Paed1986; 7: 936
6 Pless IB, Nolan T. Revision, replication andneglect research on maladjustment inchronic illness. J Child Psych Psychiatr
1991; 32: 34765
7 Diseth TH, Emblem R, Schultz A. Mentalhealth, psychosocial functioning, and
quality of life in patients with bladderexstrophy and epispadias an overview.
World J Urol1999; 17: 239488 Berg R, Berg G, Svensson J. Penile
malformation and mental health. ActPsychiatr Scand1982; 66: 398416
9 Bracka AA. A long term view ofhypospadias. Br J Plastic Surg 1989; 42:
251510 Boydell LR, Nevin NC. Is antenatal
selection for spina bifida possible? BMJ1990; 301: 608
11 Chan A, Robertson EF, Haan EA.
Prevalence of neural tube defects in SouthAustralia, 196691: effectiveness andimpact of prenatal diagnosis. BMJ1993;
307: 70312 Wolman C, Brasco DE. Factors influencing
self-esteem and self-consciousness inadolescents with spina bifida. J Adolesc
Health 1994; 15: 5438
13 Reiner S, Goldman T. A programme for theenhancement of autonomy in youngadults with physical disabilities. Int JRehab Res1999; 22: 714
14 Diseth TH, Emblem R. Somatic function,
mental health and psychosocialadjustment of adolescents with anorectalanomalies. J Ped Surg1996; 31: 63843
15 World Health Organization. Measurementof quality of life in children. Division ofMental Health, Genf, 1993
16 Bullinger M, Ravens-Sieberer U. Healthrelated quality of life assessment in
children: a review of the literature. Revue
Europeenne Psychologie Appliquee1995;
45: 24554
17 Varni JW, Seid M, Rode CA. The PedsQL:measurement model for the pediatricquality of life inventory. Med Care1999;
37: 1263918 Feeny D, Furlong W, Barr RD, Torrance
GW, Rosenbaum P, Weitzman S. Acomprehensive multiattribute system forclassifying the health status of survivors
of childhood cancer. J Clin Oncol1992;
10: 923819 Landgraf JM, Abetz LN. Functional status
and well-being of children representingthree cultural groups: Initial self-reports
using the CHQ-CF87. Psychol Health1997; 12: 83954
20 Varni JW, Katz ER, Quigging DJL,
Friedman-Bender A. The Pediatric CancerQuality of Life Inventory-32 (PQCL-32) I.Reliability and validity. Cancer, 1998; 82:
11849621 Juniper EF, Guyatt GH, Feeny DH, Ferrie PJ,
Griffith LE, Townsend M. Measuring
quality of life in children with asthma.Qual Life Res, 1996; 5: 3546
22 Wallander JL, Schmitt M, Koot HM. Qualityof life measurement in children andadolescents: issues, instruments, and
applications. J Clin Psychol2001; 57: 57185
23 Mureau MAM, Slijper FME, Nijman RJM,van der Meulen JC, Verhulst FC, Koos SlobA. Psychosexual adjustment of children
and adolescents after different types ofhypospadias repair: a norm related study.
J Urol1995; 154: 1902724 Aho MO, Tammela OKT, Somppi EMJ,
Tammela TLJ. A long term comparative
-
7/27/2019 Boala Cronica Studiu
5/5
E . W . G E R H A R Z E T A L .
1 5 4 2 0 0 3 B J U I N T E R N A T I ON A L
follow up study of voiding, sexuality andsatisfaction among men operated for
hypospadias and phimosis duringchildhood. Eur Urol2000; 37: 95101
25 Evans RL, Dingus CM, Haselkorn JK. Living
with a disability: a synthesis and critiqueof the literature on quality of life, 1985
89. Psych Reports1993; 72: 273826 Schober JM. Quality-of-life studies in
patients with ambiguous genitalia. WorldJ Urol1999; 17: 24952
Correspondence: Dr E. Gerharz, Department ofUrology, Julius Maximilians University
Medical School, Wrzburg, Germany.e-mail: [email protected]
wuerzburg.de
Abbreviations: QoL, quality of life.