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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.

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Correspondence: Dr E. Gerharz, Department ofUrology, Julius Maximilians University

Medical School, Wrzburg, Germany.e-mail: [email protected]

wuerzburg.de

Abbreviations: QoL, quality of life.

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