Self-reported quality of life of young children with conditions from early infancy: a systematic review.

Self-Reported Quality of Life of Young Children With Conditions From Early Infancy: A Systematic Review Jenni Jardine, Svetlana V. Glinianaia, Helen McConachie, Nicolas D. Embleton and Judith Rankin Pediatrics 2014;134;e1129; originally published online September 22, 2014; DOI: 10.1542/peds.2014-0352

The online version of this article, along with updated information and services, is located on the World Wide Web at: http://pediatrics.aappublications.org/content/134/4/e1129.full.html

PEDIATRICS is the official journal of the American Academy of Pediatrics. A monthly publication, it has been published continuously since 1948. PEDIATRICS is owned, published, and trademarked by the American Academy of Pediatrics, 141 Northwest Point Boulevard, Elk Grove Village, Illinois, 60007. Copyright © 2014 by the American Academy of Pediatrics. All rights reserved. Print ISSN: 0031-4005. Online ISSN: 1098-4275.

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Self-Reported Quality of Life of Young Children With Conditions From Early Infancy: A Systematic Review AUTHORS: Jenni Jardine, DClinPsy,a Svetlana V. Glinianaia, MD,b Helen McConachie, PhD,b Nicolas D. Embleton, MD,b,c and Judith Rankin, PhDb aCentral

Manchester University Hospitals NHS Foundation Trust, Salford Child and Adolescent Mental Health Service, Manchester, United Kingdom; bInstitute of Health & Society, Newcastle University, Newcastle upon Tyne, United Kingdom; and cNewcastle Hospitals NHS Foundation Trust, Newcastle Neonatal Service, Newcastle upon Tyne, United Kingdom KEY WORDS child’s self-report, congenital conditions, quality of life, systematic review, self-proxy agreement ABBREVIATIONS CHQ—Child Health Questionnaire CFQ—Cystic Fibrosis Quality of Life Questionnaire CHD—congenital heart disease DMD—Duchenne muscular dystrophy Haemo-QoL—Haemophilia Quality of Life Questionnaire HRQoL—health-related quality of life PedsQL—Pediatric Quality of Life Inventory QoL—quality of life TACQOL—TNO-AZL Child Quality of Life Questionnaire Dr Jardine performed the literature search and led the review process, reviewed all included articles, and made substantial contributions to drafting of the initial manuscript; Dr Glinianaia made substantial contributions to the study design, article reviewing, data extraction, and interpretation of data, and drafted and revised the manuscript; and Drs McConachie and Embleton made substantial contributions to the study design, article reviewing and data extraction, and critically reviewed the manuscript. Dr Rankin conceived the study; made substantial contributions to the study design, article reviewing, and data extraction; and critically reviewed and revised the manuscript. All authors approved the final manuscript as submitted. www.pediatrics.org/cgi/doi/10.1542/peds.2014-0352 doi:10.1542/peds.2014-0352 Accepted for publication Jun 25, 2014 Address correspondence to Judith Rankin, PhD, Institute of Health & Society, Newcastle University, Baddiley-Clark Building, Richardson Rd, Newcastle upon Tyne, NE2 4AX, UK. E-mail: judith. [email protected] PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 1098-4275). Copyright © 2014 by the American Academy of Pediatrics FINANCIAL DISCLOSURE: The authors have indicated they have no financial relationships relevant to this article to disclose. FUNDING: This work was supported by the United Kingdom National Institute for Health Research Newcastle upon Tyne Hospitals NHS Foundation Trust, Flexibility and Sustainability Funding.

abstract CONTEXT: There is little consistency in the use of instruments for measuring self-reported quality of life (QoL) in young children. OBJECTIVE: To systematically review studies of self-reported QoL in children aged ,12 years with congenital health conditions, and to examine the agreement between self- and proxy-reports. DATA SOURCES: Literature databases (MEDLINE, EMBASE, Web of Science, PsychINFO) were systematically searched, reference lists of eligible studies were scanned. STUDY SELECTION: We included studies published in English between January 1989 and June 2013 which used validated instruments to assess self-reported QoL in children aged ,12 years with a distinct congenital health condition identified in early infancy. DATA EXTRACTION: We extracted data on study design, objective, sample characteristics, QoL assessment instrument, statistical techniques and results. RESULTS: From 403 full-text articles assessed for eligibility, 50 studies underwent detailed review, and 37 were included in a narrative synthesis. Children’s self-reported QoL was assessed by using a variety of generic and/or condition-specific instruments, with the Pediatric Quality of Life Inventory being the most frequently used (25% [9 studies]). Regardless of the condition or the instrument used, children often reported QoL similar to the reference population, except for lower scores in the physical functioning/health domain. There were differences between younger and older age groups according to QoL domain. The child’s perception of QoL differed from that of his or her parents, in particular for subjective domains such as emotional functioning, and these differences were age related. The main limitation of the review resulted from the lack of published studies on self-reported QoL in young children, in particular, lacking both self-reports and proxy reports. Existing studies demonstrated wide variability in the QoL instruments used and approaches to statistical analyses, lack of information about the formation of the study sample (response rate; comparison of responders and nonresponders) and low sample sizes in the age group of interest. CONCLUSIONS: The reviewed studies demonstrated that, even for younger children, both child and parent perspectives are essential to understanding the impact of a condition on a child’s QoL. Pediatrics 2014;134:e1129–e1148

POTENTIAL CONFLICT OF INTEREST: The authors have indicated they have no potential conflicts of interest to disclose.

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Advances in medical treatments for many congenital and chronic childhood conditions have led to improvements in survival and reductions in key morbidities, making quality of life (QoL) an increasingly important dimension of assessment of a child’s long-term outcome.1–4 QoL is a multidimensional concept, inherently subjective, in which the self-report element for all individuals, including children, is important.5,6 Although it is well recognized in industrialized countries that ascertaining children’s views of their QoL is essential for improving health care strategies, assessment of subjective well-being in children is still not common in the United States and the United Kingdom.7 Proxy reports are often used for a child’s QoL assessment because a child is too ill or has difficulties communicating their views due to their age or because of neurocognitive impairment,8 but also because, traditionally, children were considered unreliable reporters.9 However, a number of studies have highlighted low levels of agreement between parent and child perspectives about the consequence of a condition on the child’s QoL9–11 (specifically, with poorer agreement on emotional and social domains than on physical domains9,12,13), thus implying the importance of obtaining both child and parent perspectives whenever possible.9 Several key reviews have examined studies that used QoL measures for children and adolescents.4,6,14–16 A range of issues have been highlighted: extensive variability in the content of items, limited agreement on the underlying conceptual framework,6 lack of childgenerated items,15 and lack of environmental and qualitative aspects of QoL.4 It has been concluded that it is important to differentiate QoL instruments (measuring subjective well-being [ie, happiness or life satisfaction]) from those measuring health or functioning because they are conceptually different constructs from QoL.6 e1130

The complexity of developmental and cognitive changes in childhood mean that a single instrument is unlikely to capture the varying perspectives of infants, children, and adolescents.14 Historically, self-report measures of QoL and well-being have been targeted at those aged .8 years, with proxy reports providing information for younger children.17 However, it has been demonstrated that children as young as 5 years are able to self-report their well-being.8 Although QoL instruments are now being targeted at younger age groups,18–20 there is no consensus on what constitutes a candidate measure to capture younger children’s QoL. A literature review evaluating the format and quality of health-related self-report measures for children aged 3 to 8 years identified shortcomings relating to internal reliability, validity, and responsiveness for many measures.17 Because there is little guidance regarding choice of an appropriate measure, it is important to identify the QoL instruments used by clinicians and researchers for assessment of subjective well-being of younger cohorts. The aim of the present article was to systematically review studies of selfreported QoL in children aged ,12 years with a congenital health condition and to examine the agreement between self-report and parent proxy where both were available.

METHODS Search Strategy and Selection Criteria We followed the guidance for conducting systematic reviews from the Centre for Reviews and Dissemination.21 The review was registered with the National Institute for Health Research’s PROSPERO, an international database of prospectively registered systematic reviews (CRD42012002564). Studies were identified through comprehensive literature searches of OVID Medline, Embase,

Web of Science, PsychINFO, and the Cochrane Database of Systematic Reviews. Databases were searched from January 1, 1989, to June 30, 2013; the Convention on the Rights of the Child was published in 1989,22 and it was believed that recognition for respecting the views of the child would have generated increased research for this age group. Relevant gray literature was identified by searching the NHS Evidence Database (http://evidence.nhs.uk/) and Turning Research Into Practice database (http://www.tripdatabase.com/). Reference lists of published reviews and studies were also scanned. Key words were generated through considering synonyms, undertaking pilot searches, and consulting the existing literature. Medical Subject Headings term and/or text word search strategy were used and entered systematically into the databases in the same orderand combination covering terms relating to QoL, tools/instruments, child/pediatric, and chronic health conditions topic areas (see Supplemental Appendix). The Boolean operators AND and OR were used to combine topic areas. Original studies were included if they: (1) assessed QoL of children with a distinct health condition arising in early infancy, including a congenital disease (eg, hemophilia), a structural anomaly (eg, spina bifida), or a condition resulting from events during pregnancy or in the neonatal period (eg, cerebral palsy) (for brevity, the phrase “congenital conditions” is used for the rest of the article); (2) had self-reported data on children aged ,12 years; (3) used validated generic or condition-specific QoL instruments; and (4) were published in English between January 1989 and June 2013. Studies reporting QoL in children with a diverse or mixed group of conditions of different origin were not included (eg, children with special heath care needs,23 variety of primary chronic pain conditions24). Although we acknowledge that studies of children with cerebral

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palsy will include a number of prematurely born children, we specifically excluded studies on QoL in prematurely born children because this topic was the focus of a recent systematic review.25 Studies on complications of prematurity, including chronic lung disease or vision impairment resulting from retinopathy of prematurity, were also not included. For assessment of self-proxy agreement, we applied less strict inclusion criteria for children’s age; 6 studies were included in which age groups were combined (eg, 6–13 years). Data Extraction, Quality Assessment, and Synthesis The initial reviewer (J.J.) performed all the literature searches, scanned the titles and abstracts of the identified studies (n = 8823), and reviewed all studies suitable for detailed reviewing (n = 50). Two second reviewers (S.V.G. and J.R.) also scanned the titles and abstracts of a 10% sample of the initially identified studies to confirm decisions on inclusion. The 50 studies for review were divided between 4 second reviewers by using a data extraction protocol based on a previous review.1 This method was piloted and refined before commencing the data extraction process. Articles that did not meet the inclusion criteria were excluded during the review process, resulting in 37 eligible studies (Fig 1). Data on the study design, hypothesis, sample characteristics (health condition, sample size, age), QoL instrument, statistical techniques, and results were extracted. Quality assessment was conducted by all reviewers evaluating recruitment (avoidance of selection and nonresponse bias), study design (course of measurement, informants, and type of control subjects), QoL assessment (whether validated instruments were used), and the statistical analyses (adjustment for confounders), the criteria used in previous systematic reviews.1,2 Based on these criteria, 3 quality groups were

formed: good (score 9–11), moderate (6–8), and poor (0–5). We referred to the PRISMA (Preferred Reporting Items for Systematic Reviews and MetaAnalyses) standards throughout the process of conducting and writing this systematic review (http://www.prismastatement.org/statement.htm). Because of the varied QoL measurements and summary measures, it was not possible to conduct a meta-analysis; the results were therefore synthesized narratively. Studies rated as poor quality were accorded less weight when drawing conclusions.

RESULTS Overview of Included Studies The initial searches identified 9998 publications; after removing duplicates (n = 1178), the studies were screened by title and abstract. Due to the limited information within the abstracts regarding the age range, origin of the condition, and uncertainty relating to self-report, a substantial number of articles (n = 403) underwent a full review. The reasons for exclusions at this phase are listed in Fig 1. Table 1 presents a list of validated QoL instruments used by the 37 included studies to assess the subjective QoL of children aged ,12 years and parent proxy. Ten self-report generic QoL instruments and 5 condition-specific instruments were identified; 5 studies used measures of psychological adjustment, resulting in heterogeneity in the QoL domains included. The Pediatric Quality of Life Inventory (PedsQL) for 2- to 18-year-olds was used most frequently (n = 9). Many of the studies had methodologic limitations: they were derived from convenience samples; were of crosssectional design with a low or unreported response rate; and included no description of nonresponders. About one-half of the studies focused on a single informant only; in some studies with

both parent and self-reports, however, it was not always possible to assess agreement because children and parents completed different outcome measures.26 All these limitations affected the study quality, which was scored as moderate for most studies. Studies of Self-Reported QoL Table 2 provides details of 21 studies for children aged ,12 years. Some studies included older children in their sample but separated results for the younger cohort.27–34 The studies covered a variety of conditions, including congenital heart disease (CHD) (n = 7), cerebral palsy (n = 2), hemophilia (n = 2), and orofacial clefts (n = 2). The majority of studies either used reference data or a control group for comparison; the remaining studies used within-group age comparisons27,28,31 or compared one congenital condition with a related condition.35 Overall, irrespective of the condition or the instrument used, children often reported similar scores for their QoL compared with the reference scores. For example, in one study, “school environment” was the only QoL domain reported to be significantly better in children with cerebral palsy according to the KIDSCREEN questionnaire.36 Another study demonstrated that although children with cerebral palsy reported lower health status than control children (measured by using the Child Health Questionnaire [CHQ]–Child Form), reflecting functional levels of activity, functional abilities/impairments did not reduce the children’s self-reported QoL in any of 5 domains measured by using the Youth Quality of Life Questionnaire compared with the matched control subjects.37 Similarly, QoL was generally good in boys with hemophilia; however, there were noticeable differences in QoL impairments according to domain between younger and older groups when



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FIGURE 1 Flowchart of selection of articles for systematic review.

measured by using either the diseasespecific instrument (Haemophilia Quality of Life Questionnaire [Haemo-QoL])27,28 or the generic instrument (KINDL).28 Thus, boys aged 8 to 12 years reported worse total scores on both the KINDL and Haemo-QoL questionnaires than younger boys, and they were particularly vulnerable in “self-esteem” and “school” domains (KINDL) and “friends,” “dealing,” “support,” and “treatment” domains (Haemo-QoL)28 (Table 2).

(aged 5–7 years), self-perceived QoL (measured by using KINDL) was even better than in same-aged peers from the general population.40 In contrast, in a study using PedsQL, for both 5- to 7year-olds and 8- to 12-year-olds, QoL in children with CHD was found to be significantly lower in all domains compared with the age-matched reference group.33 Self-reported QoL in 8- to 12year olds in another similar study was comparable with control subjects.34

Studies using various tools (Self-Concept Scale, KINDL, and the Lindström method) in children with CHD38–40 also reported QoL in most domains comparable with the reference group, except for physical self-concept.39 For younger children

Younger children (aged 8–10 years) with orofacial clefts had a significantly higher average global health–oral score than the 15- to 18-year-olds.30 However, there were no significant differences between younger (6–11 years) and older

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($14 years) age groups of patients with cystic fibrosis in all domains of selfreported QoL measured by using the Cystic Fibrosis Questionnaire (CFQ), except “treatment.”31 In other studies, children with sickle cell disease, spinabifida,Duchennemuscular dystrophy (DMD), bladder exstrophy, lower urinary tract dysfunction, and congenital hearing impairment reported impaired QoL compared with the general population or age-matched control children (Table 2).29,32,41–44 Perceived QoL in boys with DMD measured by using PedsQL was lower for all domains, except for the emotional domain, compared with unaffected age-matched control subjects.32 However, for boys

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TABLE 1 Instruments for Assessment of QoL Used by the Included Studies QoL Assessment Instrument

No of Studies/Reference

Psychological adjustment Child Behavior Checklist (proxy report) Children’s Depression Inventory (self-report) Primary Self-Concept Inventory (self-report) Self-Perception Profile for Children (self-report) Self-Concept Scale (self-report) State-Trait Anxiety Inventory for Children (self-report) Youth Self-Report (self-report) HRQoL (generic) Autoquestionnaire Qualité de Vie Enfant Imagé, Quality of life evaluation scale for children aged 6–12 y CHQ–Child Form 87 (10–18 y) and CHQ–Parent Form 28 (5–18 y) KIDSCREEN (8–18 y) KINDL (has age-specific versions for self-reported QoL [eg, Kiddy-KINDL (for children aged 4–6 y), child version]) Lindström method, validated instrument measuring 3 life spheres of external living conditions and interpersonal and personal conditions64,65 TACQOL–Parent Form (for children aged 6–11 y) and TACQOL–Child Form (for children aged 8–15 y) PedsQL for children aged 2–18 y, child and parent versions Vécu et Santé Perçue de l’Enfant for children aged 12–17 y Youth Quality of Life Questionnaire Condition-specific HRQoL Child Oral Health Impact Profile Cerebral Palsy Quality of Life Questionnaire for Children for 9- to 11-year-olds (child’s self-report) and for 4- to 12-year-olds (parent proxy version) CFQ Haemo-QoL Hirschsprung Disease/Anorectal Malformation Quality of Life Questionnaire

3/26, 40, 57 1/26 1/63 1/26 1/39 1/26 1/57 2/42, 44 2/37, 51 4/11, 36, 41, 53 6/28, 29, 40, 54–56 1/38

3/35, 46, 58 9/32–34, 43, 45, 48–50, 59 1/42 1/37 1/30 2/52, 53

4/31, 47, 48, 60 2/27, 28 1/35

Some studies have used .1 QoL measure.

aged ,10 years, QoL for social, emotional, or school domains was comparable to control subjects. Interestingly, self-reported QoL was not different between younger (,10 years) and older ($10 years) boys with DMD, despite the significant decline in engagement in social activities for older boys. In another study, boys with DMD also reported significantly higher scores for physical, but not psychosocial functioning, in younger (8–12 years) than older (13–17 years) age group.45 Studies Comparing Self-Reports and Proxy Reports

sample size and in the approach to data analysis and presentation. Ten studies analyzed the level of agreement between child and parent scores through correlations11,33,40,45–51 (Pearson or Spearman rank correlation or intraclass correlation coefficients), whereas 11 used other approaches in which there was no statistical analysis of self-proxy agreement reported31,34,52–60 (Table 3). Only 1 study analyzed the magnitude of the disagreement or agreement, taking into account the average SDs of the child and parent scores (effect size), the direction of self-proxy difference, and the factors affecting it.11

We identified 21 studies that collected QoL data from both children and parents (5 of 21 are also presented in Table 231,33,34,40,46). There was substantial variation between these studies in

Of the 21 studies, the most common conditions were as follows: cerebral palsy (n = 5), cystic fibrosis (n = 4), and CHD (n = 4). Most studies used generic QoL questionnaires that have both

parent and child versions (ie, PedsQL [n = 7], KINDL [n = 4], KIDSCREEN [n = 2], TNO-AZL Children’s Quality of Life Questionnaire [TACQOL; n = 2], and CHQ [n = 1]). In several studies, disease-specific measures (ie, the Cerebral Palsy Quality of Life Questionnaire for Children [n = 2] and CFQ [n = 4]) were used individually or in addition to generic instruments. The majority of these studies reported that a child’s perception of QoL differed from that of his or her parents, with parents frequently underestimating their children’s QoL,11,49,56 in particular in subjective domains (eg, emotional and psychosocial).34,45,47,49,51,52,58,59 Despite reports of weak correlation11,47,48 or large differences in mean scores58 between selfreports and proxy reports for the emotional domain, White-Koning et al11 demonstrated that the self-report–proxy agreement rate for “emotions” was higher (44%) than for most domains (range: 25%–40%). Nevertheless, some studies found that parents overestimated the QoL of their children; for example, for positive emotions in children with congenital hypothyroidism58 or for all domains for 5- to 7-year-olds with CHD.33 Interestingly, using the same QoL instrument (KINDL), 8- to 12-year-old children with hearing impairment rated their overall QoL significantly lower than their parents did in one study,55 whereas in another study, hearing-impaired younger children (aged 4–7 years) reported significantly better QoL than parents did on the overall score.54 Overall, studies using either a more functioning-oriented instrument (eg, CHQ, PedsQL, TACQOL) or a more well-being–oriented instrument (eg, KIDSCREEN, KINDL) found discrepancies between self-reports and proxy reports. However, the direction of discrepancies (parent or child rating better QoL), as well as disagreement for specific QoL domains, was inconsistent across studies. When it was possible to compare the results between different age groups



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Hemophilia Gringeri et al (2004),27 6 West European countries; multicenter

Duchenne muscular dystrophy Bendixen et al (2012),32 United States

Bjornson et al (2008),37 United States

Cerebral palsy Dickinson et al (2007),36 6 European countries

Author, Location, Year

95 (4–7 y) 118 (8–11 y) 105 (13–16 y)

50 (,10 y = 27) ($10 y = 23)

81

500

Sample Size

4–16; (4–7); (8–11)

5–15 (,10); Mean: 8.9 6 2.4

10–13

8–12

Age of Child (y) (Range or Mean)

94% (318 from 339 enrolled)

Not stated

56% (81 of eligible 145)

61% capable of selfreporting QoL

Response Rate (%)

TABLE 2 Details of the Included Studies of Self-Reported QoL

Age group comparison

Control group: unaffected agematched boys (n = 25; ,10 y, n = 13)

Control group: age- and gender-matched children with typical development (n = 30)

General population norms: mean scores for KIDSCREEN

Comparison Group

Haemo-Qol (higher values = higher impairment)

CAPE (measure of participation)

PedsQL

CHQ-CF87

YQOL-R

KIDSCREEN

Instruments Used

HRQoL deemed satisfactory in this cohort with the most mean scores much lower 50. Young children (4–7 years) impaired in “family” (34.4 6 24.8) and “treatment” dimensions. Children 8 to 12 years old reported worse scores in social dimensions of “perceived support” (49.4 6 25.2) and “friends” (43.9 6 28.05).

Significantly lower scores for ,10-year- old DMD children versus control children only for “total” score and “physical” QoL subscale but not “social,” “emotional,” or “school.” Older boys also had lower scores on social subscale. For all ages combined, QoL was lower in boys with DMD in 3 domains and total score, except “emotional.” No differences in QoL between ,10- and $10-year-olds with DMD. Participation in physical activity significantly lower in patient group. Over time, the amount of time engaged in activity and social activity declined in older boys but did not affect QoL.

Children with CP had similar QoL to children in the general population on all domains except “schooling,” in which the scores were significantly better. Specific impairments were associated with poorer QoL: poor walking ability and poor “physical well-being”; greater intellectual impairment had lower “mood and emotions” and less “autonomy”; and those with speech difficulties had poorer “relationships with their parents.” Self-reported QoL did not significantly differ in any of 5 domains in children with CP versus control children irrespective of their activity levels. Youth with CP reported significantly lower health status in the following subscales: physical function, general health perception, bodily pain, role/social physical, and role/ social behavioral. There were significant differences across GMFCS levels.

Main Findings According to Authors

Good

Poor

Moderate

Good

Study Quality

Møyen Laane et al (1997),38 Norway and Sweden

Hövels-Gürich et al (2007),40 Germany

CHD Chen et al (2005),39 Taiwan

Khair et al (2012),28 United Kingdom


TABLE 2 Continued

164

2–12; Median: 6 y, 1 mo

5–7 (n = 28); 8–12 (n = 12)

20 TOF,

20 VSD

5.0–11.8

40

8.6–12.7

6–17 (6–7; 8–12)

84: 15 (6–7 y), 41 (8–12 y)

64


Sample Size

75% of control subjects

82%

Not stated

Not stated, convenience sample

70%

Response Rate (%)

Control group of agematched unaffected children (n = 301)

Two study groups: VSD and TOF

Published norms of German school pupils

Comparable control group (n = 71) of unaffected children

Age group comparison, and between those doing sports versus those not doing sports

Comparison Group

Lindström method

CBCL

KINDL (Kiddy-KINDL for 5–7 y; KidKINDL for 8–12 y)

SCS

Haemo-QoL (higher values = higher impairment)

KINDL questionnaire (lower values = higher impairment)

Instruments Used

Significantly lower scores on physical SC (P = .007) but not for family SC, school SC, appearance SC, and emotional and general SC. Scores for physical SC remained significantly lower in patients versus control subjects after adjustment for confounders. Children 5 to 7 years old reported better QoL (total score) than the same age reference group. Self-reported QoL scores for 8- to 12-year-olds did not differ from those in control subjects in any domain. Between groups of children with TOF and VSD, the only difference was found on “friend” domain, which was rated higher in VSD group (P = .04). Internalizing and externalizing difficulties elevated. School performance and total competence reduced. No difference between the CHD groups and control subjects for a total mean in any of the 3 spheres (“external,” “internal,” and “personal”). However, in the personal sphere, the total CHD group had a lower score for peeracceptance than the control subjects mainly due to lower scores in the groups treated by surgery and with associated anomalies (P , .05). Self-esteem was reported to be significantly lower in the CHD group with associated anomalies versus control subjects (P , .05). In some domains (lack of psychosomatic problems, satisfaction with social support), scores were higher in CHD groups than in control subjects.

The highest impairments (KINDL) in the 8-to 12-year-old group were in the dimension “school” (55.01 6 17.2) and selfesteem (59.5 6 17.1), whereas scores for 6- to 7-year-olds were much higher for these dimensions (75.0 6 31.0 and 75.0 6 23.1, respectively). HRQoL in boys with hemophilia was generally good, with highest impairments in disease-specific HRQoL in boys aged 8 to 12 years (Haemo-QoL total mean = 41.43 6 10.5). Children 6 to 7 years old reported worse scores in “sport and school,” whereas in 8- to 12-year-olds, “dealing” (with the disease), “friends,” “treatment,” and “support” dimensions were mostly affected.


Moderate

Moderate

Moderate

Moderate

Study Quality

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8+ y (n = 23)

Orofacial clefts Broder and Strauss (1989),63 United States

40

6–9

Anorectal anomaly or Hirschsprung’s disease Hartman et al 250 (8–16) 8–11 (n = 141); (2007),35 the 12–16 (n = 109) Netherlands; longitudinal

23 self-report


84% for 8- to 11-yearolds Compared responders and nonresponders

35% (57 of 163)

Mean: 8 y 8 mo 6 1 y 6 mo

43

Miatton et al (2007),26 Belgiuma

73%

8–15 (8–11; 12–15)

113: 58 (8–11y)

Spijkerboer et al (2006),46 the Netherlands

Convenience sample (recruited from pediatric cardiology unit)

475 of 500 families, but 347 child responders

Convenience sample

Response Rate (%)

2–18; 5–7; 8–12

5–7, 8–12, 13–18

5–18;


195 (5–18); 38 (5–7); 63 (8–12)

347: 65 (5–7); 142 (8–12); 140 (13–18)

Sample Size

Berkes et al (2010),34 Hungary

Uzark et al (2008),33 United States


TABLE 2 Continued

Control group of sexmatched children of a similar age; groups with cleft lip, cleft lip/ palate, and cleft palate were also compared

Anorectal anomaly compared with HD

Controls matched by age, gender, education of both parents and educational level of the child

Control subjects randomly selected from the general population (n = 525 families; response rate: 52.5% [373 children]) Comparable age distribution Dutch normative samples from the general population for 8- to 11-year-olds and 12- to 15-yearolds

Healthy child norms for the same age groups

Comparison Group

PSCI

HAQL

TACQOL

STAI

CDI

SPPC

TACQOL-child

Cardiac module of PedsQL

Generic Core Scales

PedsQL

PedsQL

Instruments Used

Children with both cleft lip and palate had the lowest selfconcept score compared with control subjects and other cleft defects (cleft lip only and cleft palate only).

Children with both conditions improved in disease-specific functioning (P , .01) and mental QoL (P = .01) over a 3-year period. Those with a severe form of HD or with additional congenital anomalies exhibited worsening of “school attitude” that affected change in “mental QoL” negatively.

Compared with same-aged healthy peers, 8- to 11-year-old children with CHD reported significantly lower (P , .05) mean scores on 5 of the 7 TACQOL scales: motor functioning, autonomy, cognitive functioning, social functioning, and positive emotional functioning. No significant differences on the scales “pain and physical symptoms” and “negative emotional functioning.” No significant difference from control subjects for scholar competence, social acceptance, athletic skills, physical appearance, behavior, and self-worth. More depressive feelings than healthy control subjects (P = .007). State anxiety; not significant difference from control subjects.

For both groups (5–7 years old and 8–12 years old), QoL in all domains was significantly lower compared with the reference group. 5–7 years: Total score = 71.9 vs normal score = 82.2 (P , .001). 8–12 years: Total score = 78.1 vs normal score = 84.4 (P , .001) In 5- to 7-year-old group, children reported significantly lower HRQoL in all domains compared with healthy control subjects, except for emotional functioning. In 8- to 12-year-old group, children’s QoL scores were comparable to control subjects in all domains. Compared with children with simple CHD, total, physical, and social functioning scores were lower in patients with CHD of great complexity (P , .05).


Poor

Moderate

Moderate

Moderate

Good

Moderate

Study Quality

50 33 (8–12 y) 17 (13–16)

Lower urinary tract dysfunction Lopes et al 28 (2011),44 Brazil

Congenital hearing impairment Rajendran and 100 (group 2) Roy (2010),43 100 (group 3) India

Spina bifida Müller-Godeffroy et al (2008),29 Germany

40

75 22 (8–10) 35 (11–14) 18 (15–18)

Ward et al (2013),30 United Statesb

Sickle cell disease Hijmans et al (2010),41 the Netherlands

Sample Size


TABLE 2 Continued

4–12 4–7 (n = 5), 8–12 (n = 23)

6–11

6–16 y 8–12

6–11 (n = 17) vs healthy siblings (n = 19)

8–18



Not stated

62%

87% (40 of 46 randomly selected)

93.8% (75 of 80)

Response Rate (%)

Control group (n = 38) of typically developing children

Age-matched healthy controls (n = 100, group 1)

General population norms for KINDL

Control group of healthy siblings and Dutch norms

75 control subjects

Comparison Group

AUQUEI

PedsQL

KINDL

KIDSCREEN-52

COHIP, 38-item questionnaire

Instruments Used

Patients had ,48 points (low QoL) in total AUQUEI scoring in 32% versus 8% in control subjects (P , .0213), and they also had problems in dealing with social aspects, such as being in the classroom.

Significantly lower HRQoL for hearing-impaired children with motor impairment (group 3) on all domains (P , .001). Children with hearing impairment and no motor impairment (group 2) had significantly lower scores in emotional health and school function domains but not in physical and social health domains.

Children with spina bifida (8–12 y) reported lower HRQoL in all dimensions (“emotional,” “self-esteem,” and “friends”) and total score (medium to large effect sizes). Adolescents reported lower scores on peer relations.

HRQoL in 6- to 11-year-olds was comparable to healthy siblings but significantly worse compared with Dutch norms on 7 of 10 domains: “physical well-being,” “moods and emotions,” “self-perception,” “autonomy,” “parent relation,” “finances,” and “bullying.” Healthy siblings had worse QoL than Dutch norms on “autonomy,” “parent relation,” and “finances.” In 12- to 18-year-olds, only “autonomy” scores were significantly lower compared with Dutch norms.

For the whole group (8–18 years of age), children with orofacial clefts had lower QoL scores than control subjects for overall oral health-related QoL, functional well-being, and social-emotional well-being. However, there were age-related differences; for example, for social-emotional well-being, control subjects had a higher average score (P = .017) than the cleft group only for 15- to 18-year-olds. The 8- to 10-year-old group had a higher average score (global health–oral) than the 15- to 18-yearold group (P = .039).


Moderate

Poor

Moderate

Moderate but n = 17

Moderate

Study Quality

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Mean 7.7 6 1.6; children n = 17

6–26: (6–11, n = 39) (12–13, n = 12)

75

71 (whole sample including adults)


Sample Size

53%

Not stated

Response Rate (%)

VSP-AE

AUQUEI

French population norms

CFQ

Instruments Used

Patients with bladder exstrophy – age group comparison with adolescents

Patients with cystic fibrosis–age group comparison

Comparison Group

Moderate, but low number of children

Children’s QoL globally lower than the general population except for “relations with family” and “school work.”

Among patients, children’s scores were higher than adolescents on all VSP-AE dimensions (and global score), except for “self-esteem,” “vitality,” and “relations with friends.” Authors do not report results from AUQUIE

Moderate

Study Quality

No significant difference between age groups (6–11 years; 12–13 years; .14 years) for all domains except for “treatment” between 6- to 11-year-olds and $14-year-olds (P = .001).


AUQUEI, Quality of life evaluation scale for children aged 6 to 12 y (Autoquestionnaire Qualité de Vie Enfant Imagé); CAPE, Children’s Assessment of Participation and Enjoyment; CBCL, Child Behavior Checklist; CDI, Children’s Depression Inventory; CHQ-CF87, Child Health Questionnaire–Child Form 87; COHIP, Child Oral Health Impact Profile; CP, cerebral palsy; DMD, Duchenne muscular dystrophy; GMFCS, Gross Motor Function Classification System; HD, Hirschsprung’s disease; Haemo-Qol, Haemophilia-specific QoL Questionnaire; HAQL, Hirschsprung Disease/Anorectal Malformation Quality of Life Questionnaire; PSCI, Primary Self-Concept Inventory; SC, self-concept; SCS, Self-Concept Scale; SPPC, Self-Perception Profile for Children; STAI, State-Trait Anxiety Inventory for Children; TOF, tetralogy of Fallot; VSD, ventricular septal defect; VSP-AE, Vécu et Santé Perçue de l’Enfant; YQOL-R, Youth Quality of Life Questionnaire–Research. a Both self-report and parent proxy report were used, but the outcome measures were different; hence, it was not possible to assess the self-report–proxy agreement and is therefore not included in Table 3. b Both self-report and parent proxy report were used, but child and parent comparisons were not separated by age group; they are given for the whole 8- to 18-year-old group and therefore not included in Table 3.

Jochault-Ritz et al (2010),42 France

Bladder exstrophy

Cystic fibrosis Cohen et al (2011),31 Brazil


TABLE 2 Continued

within the same study, the differences were found to be age related. For example, in a study of children with CHD,34 there was more agreement between 5to 7-year-old children and their parents in which both self-reports and proxy reports rated QoL lower than healthy control children for all domains, except for emotional functioning in self-report. For 8- to 12-year-olds, parents scored the QoL of their children lower than children themselves in all domains.34 In contrast, another study using the same instrument (PedsQL) found low selfreport–proxy agreement for 5- to 7-yearolds with CHD but better agreement for 8- to 12-year-olds.33

DISCUSSION

This systematic review identified 37 studies that measured self-reported QoL in children aged ,12 years with a congenital health condition. The findings of the reviewed studies confirm the importance of obtaining children’s own views on their QoL and distinguishing their views by age group. Overall, irrespective of the condition or the QoL instrument used, children often reported QoL scores comparable to the reference scores, except for the physical functioning/ health domain for some conditions. Depending on the condition, younger children usually reported better overall QoL, but there were significant differences in QoL impairments according to domain between younger and older age groups. The few studies available on young children found that children as young as 4 years of age were able to report their QoL if assisted by specifically designed questionnaires with simplified response scales or an interview.

Studies that considered agreement between child-self and parent-proxy reports found that children’s perception of QoL differed from that of their parents, in particular for subjective domains, such as emotional domain, and these differences were age related.

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95

500

72

White-Koning et al (2007),11 European study involving 7 countries

Davis et al (2009),53 Australia; RCT (therapeutic horse riding, 10-wk program)

6

No. of Child–Proxy Pairs

Majnemer et al (2007),49 Canada

Cerebral palsy Dieruf et al (2009),50 United States; intervention pilot study

Author, Location


Downloaded from pediatrics.aappublications.org at Eccles Health Sciences Lib on November 29, 2014 Child – 9–12y

Both proxy and child – for 9– 12 y (n = 21)

Proxy – 4–12y CP-QoL-Child Proxy for 4–12y; child version for 9–12y

KIDSCREEN-10

KIDSCREEN

PedsQL

PedsQL

QoL Instrument

4–12

8–12

5–12

6–14

Age Range (y)

Summary of Findings

Preintervention and postintervention (intensive Poor to moderate agreement between weight-supported treadmill training) parent proxy and child self-report. Total score pre-ICC = 0.32 Authors attribute some illogical Total score post ICC = 0.56 scores (low or negative) to small Psychosocial pre ICC = 0.38 sample size. Psychosocial post ICC = 0.64 Physical pre ICC = 0.56 Physical post ICC = 0.61 (No P value given) Physical well-being r = 0.59, P , .0001 Children rated their QoL higher than their Psychosocial well-being r = 0.39, P = .01 parents, in particular for psychosocial well-being. Social acceptance: ICC = 0.32, directional Correlation between children and difference (DD) = 3.3 (622.0), P = .001 parents scores low. Parental relations: ICC= 0.21, DD = 6.0 (620.0) In 8 of 10 domains, children reported on (P , .0001) average better QoL than parents (moderate effect in most domains). Self-perception: ICC = 0.23, DD = 4.5 (621.0) Parents rated child’s Qol lower except for P , .0001 “finances.” No significant difference on “emotional” domain. Psychological well-being: ICC = 0.22, DD = 7.5 The average frequency of disagreement (618.7) P , .0001 over all domains was 64%, with parents rating their child’s QoL lower than their children in 29% to 57% of child–parent pairs. School: ICC = 0.41, DD = 4.8 (620.2) P , .0001 Range of rates of agreement was 25% to Autonomy: ICC = 0.22, DD = 7.4 (623.9), P , .0001 40% in most domains, being highest Physical well-being: ICC = 0.28, DD = 9.9 (623.0), for emotions (44%) and social P , .0001 acceptance (55%). Social support: ICC = 0.25, DD = 15.4 (624.6), P , .0001 Finances: ICC = 0.38, DD = 23.4 (632.8), P = .04. Emotions: ICC = 0.24, DD = 1.1 (618.8), P . .05 At time 2 (after the intervention) there was slight Therapeutic horse riding does not have difference in parent-proxy global HRQoL a clinically significant impact on the scores (P = .04) in contrast to no difference in QoL of children with CP. child self-report (P = .74) No difference in CP-QoL scores after the intervention by either parent or child selfreport.

Comparison of Child and Proxy Reports (Interrater Agreement Where Available)

TABLE 3 Details of Included Studies of Self-Reported and Proxy-Reported QoL: Agreement Between Child and Parent Ratings

Good

Good

Moderate

Pilot study

NA

Study Quality

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Havermans et al (2006),47 Belgium

Cystic fibrosis Thomas et al (2006),48 Australia

Shelly et al (2008),52 Australia

Author, Location

TABLE 3 Continued

36

156

205 parents of 4–12 y 53 children aged 9–12 y


CFQ PedsQL

CP-QoL

QoL Instrument

Summary of Findings

Study Quality

Parent proxy (7 domains) For parent report, functioning level was Moderate All domains of QoL were significantly associated significantly associated with all with functioning, except “access to services.” domains of QoL, whereas for child selfChild self-report (5 domains) report, it was not associated with “Feelings about functioning,” “participation and either emotional well-being or social physical health,” and “pain and feelings about well-being and acceptance. disability” were significantly associated with level of functioning. There was no association with “emotional well-being” or “social wellbeing and acceptance.”


Pearson correlation: Strong correlations between parent Moderate Physical r = 0.70, P , .001 proxy and child self-report for Body image r = 0.43, P , .001 physical, body image, eating, and Eating r = 0.60, P , .001 respiratory domains. Weaker Respiratory r = 0.63, P , .001 correlation on emotions. Emotion r = 0.24, P = .036 Significant correlation between all Treatment burden r = 0.28, P = .013 PedsQL parent and child scores Digestion r = 0.29, P = .01 Parents scored significantly lower for physical, body image, eating (P = .01) and treatment burden (P , .001, paired t tests) Physical r = 0.61, P , .001 Emotional r = 0.52, P , .001 Social r = 0.53, P , .001 School r = 0.57, P , .001 Psychosocial summary r = 0.65, P , .001 Total scale score r = 0.67, P , .001 Children aged 5 to 7 y had a lower social HRQoL score compared with parent proxy (P = .03, paired t test). 6–13; mean 9.3 6 CFQ Respiratory symptoms ICC = 0.50 Except for ratings on respiratory and Moderate 2.2 6–11 by interview Digestive symptoms ICC = 0.41 digestive symptoms, children’s scores 12–13 y (n = 7) self- Body image ICC = 0.40 were higher. In particular for completion Eating disturbances ICC = 0.75 emotional functioning and treatment Emotional functioning ICC = 0.01 burden, they reported much higher Treatment burden ICC = 0.18 QoL than their parents. Physical symptoms ICC = 0.01 Paired t test: Children rated their QoL significantly higher than their parents on emotional functioning (P , .001), treatment burden (P , .001), and body image (,0.01) and lower for respiratory symptoms (P , .01).

5–7 (n = 27) 8–12 (n = 48)

4–12

Age Range (y)

Uzark et al (2008),33 United States

475 parents 347 children

40 20 – VSD 20- TOF

35

DMD Bray et al (2010),45 Australia

Congenital heart disease Hövels-Gürich et al (2007),40 Germany

51

33


Cohen et al (2011),31 Brazil

Hegarty et al (2009),60 Australia

Author, Location

TABLE 3 Continued Summary of Findings

Study Quality

Treatment burden: child mean = 71.72 (SD = 25.1), Young people perceive the “burden of Moderate parent mean = 48.89 (SD = 28.5); F1,40 = 7.615, treatment” on their daily life less than P = .009. their parents. No difference in scores between young people and parents across other domains (eg, “emotional state” and “body image”). Details on statistical analysis of agreement not Good agreement was established between Moderate reported. the mean scores of CFQparents 6–13 and Significantly higher scores reported by parents child self-report. However, for 6- to of 6- to 11-year-olds than 12- to 13-year-olds on 11-year-olds, parents tended to the emotional domain only (P = .043), whereas report higher scores than children no differences found by self-report. for emotional and social domain.


5–18; PedsQL 5–7 (n = 65), 8–12 (n = 142)

Correlation of KINDL total scores for all patients After corrective cardiac surgery, selfModerate (Spearman: 0.43; P = .006) and subgroup of 8reports and parent-reported total to 12-year-olds (Spearman: 0.72; P = .008) scores significantly corresponded in all patients as well as in subgroup of 8to 12-year-olds. Moderate Overall, scores (self-report versus proxy) were Although self-reported QoL scores for similar for total scores (78.1 vs 78.0), physical both 5- to 7-year-old and 8- to 12-yearhealth (81.9 vs 83.3), emotional (73.9 vs 73.6), old CHD patients were significantly and social functioning (80.3 vs 80.4) in 8- to 12lower in all domains compared with year-olds. healthy peers, proxy reports were Agreement was low for 5- to 7-year-olds. similar to norm proxy reports for both age groups except for “emotional functioning” (P , .001), “psychosocial health,” and “school functioning” (P , .01) in 8- to 12-yearolds. ICC comparisons within patient group by disease Symptom scale mean scores were severity category: total sample was 0.41 to significantly related to severity 0.61, highest for physical functioning and category by both parent and selfcardiac symptoms. report.

PedsQL for 8–12 and Physical functioning ICC = 0.51; 95% CI: 0.08–0.75; Parents underestimate their child’s Moderate, but age 13–18 y r = 0.65 (P , .01). HRQoL with poor to moderate correlation on dimensions. combined for Emotional functioning ICC = 0.43; 95% CI: 0.12– Self-report: significantly higher scores assessing 0.66; r = 0.47 (P , .01). for physical (P = .0001) but not self-proxy psychosocial functioning for 8–12 y vs agreement School functioning ICC = 0.64; 95% CI: 0.40–0.80; 13–17 y groups. r = 0.66 (P , .01) Social functioning ICC = 0.20; 95% CI:20.08 to 0.47; r = 0.26 (P $ .05)

CFQ

CFQ

QoL Instrument

5.0–11.8: 5–7 y, 8– KINDL 12 y

13–17

8–12

Mean 12.5 6 2.8

9–17;

12–13

6–11

14–18 (n = 11)

6–13 (n = 22)

Age Range (y)

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38 (5–7 y) 63 (8–12 y)


Congenital hearing impairment Warner-Czyz et al (2009),54 50 children United States 45 pairs

Spijkerboer et al (2006),46 Netherlands

Berkes et al (2010),34 Hungary

Author, Location

TABLE 3 Continued

4–7

8–15 8–11 (n = 58) 12–15

2–18 5–7; 8–12

Age Range (y)

Kiddy-KINDL Interview format

TACQOL-CF TACQOL-PF

PedsQL Cardiac module of PedsQL (age collated by severity and diagnostic group)

Cardiac module of PedsQL

QoL Instrument

Study Quality

Moderate

Moderate Children (aged 8–15 y) reported significantly poorer QoL on 4 of 7 scales compared with parental report: pain and physical symptoms (P , .001), motor functioning (P , .001), autonomy (P = .006), and positive emotional functioning (P = .001). The mean disagreement between selfreport and proxy reports was largest for pain and physical symptoms for 8to 11-year-olds. The difference between child and parent reports of children aged 8–11 years for positive emotional functioning was larger than that for older children.

In the 5- to 7-year-old group, both the Good child and parent reported significantly lower HRQoL in all domains compared with healthy control subjects, except for emotional functioning in self-report. Parents reported lower scores in all domains for 8- to 12-year-olds and in “emotional functioning” for 5- to 7year-olds than children themselves.

Summary of Findings

Paired t test: Children with CHI reported scores for Children with cochlear implants scored overall QoL similar to children with significantly higher than parents on overall normal hearing. QoL (MeanDifference = 4.22, SDDifference = 12.45, Children reported significantly better P = .03). QoL than parents on overall score.

By parent report, psychosocial health scores decreased incrementally as severity increased (P , .01); by self-report, differences in psychosocial health scores across disease severity categories were less significant. Patient group (median scores); control group (mean scores) 5–7 y: Child total = 70.65 vs controls total = 79.20 (P = .0007) Proxy total = 72.83 vs control proxy report total = 77.95 (P = .0108) 8–12 y: Child total = 80.43 vs controls total = 79.47 (P = .21). No significant differences from control subjects in individual domain scores. Proxy total = 76.09 vs control proxy report total = 78.3 (P = .021). Significant differences from control subjects only in “physical health” domain (P = .005). Pearson correlation: For all TACQOL scales (pain and physical symptoms, motor functioning, autonomy, cognitive functioning, social functioning, positive emotional functioning, and negative emotional functioning) correlations were significant at P , .01; however, the correlation was large only for motor functioning (r . 0.50). Coefficient (r) range: 0.26–0.51 Pain and physical symptoms, social functioning, and positive emotional functioning: r “medium” (0.30–0.49).


Orofacial clefts Kramer et al (2008),56 Germany

Panepinto et al (2009),59 United States

Sickle cell disease Panepinto et al (2005),51 United States

Huber et al (2005),55 Austria; pilot study

Author, Location

TABLE 3 Continued

147

40 14 (5–7 y) 26 (8–12 y)

95 parents; 53 children; 49 pairs

44 patients with cochlear implants


5–6

2–18

5–18

Final n = 18

8–12 (n = 30)

8–16

Age Range (y)

KINDLR

Summary of Findings

Study Quality

The scores of children were much higher than Overall, the impact of orofacial clefts on Moderate those reported by parents in all 6 dimensions QoL was low. (P , .001). Children rated their QoL better than their parents.

According to parents (but not children), children with severe SCD also have increased odds of worse total QoL score and psychosocial health independent of covariates (child’s age, family income, and comorbidities) compared with healthy children.

Both parent proxy and child reports Moderate but showed that severe SCD is associated age with significantly higher risk of worse combined HRQoL for “physical health.”

Parents underestimated children’s QoL, Moderate, but reporting worse HRQoL in the general age health perception, physical combined functioning, behavior, and self10–18 y esteem domains (P , .005 for each). Disease status, neurobehavioral comorbidities and parent education associated with HRQoL of the child.

Parents rated their child’s QoL more positively for “physical well-being” but less positively for other domains (no significance tests conducted). Child HRQoL total 64.6 (SD 8.9) vs Norm HRQoL 8- to 12-year-olds report significantly Moderate total 76.8 (SD 8.6); P , .001 lower score on total HRQoL score compared with hearing children. Parent HRQoL total 80.8 (SD 5.4) Parents rate QoL of their children much higher. Paired difference between self-report and proxy report: mean 16.29 (SD 10.95); P , .0001.


Pearson correlation: Physical functioning r = 0.44 (P , .005), DD = 216.4 (P , .005) Bodily pain r = 0.58 (P , .005), DD = 24.29 (P $ .05) Behavior r = 0.45 (P , .005), DD = 215.2 (P , .005) General health perception r = 0.44 (P , .005), DD = 217.24 (P , .005) Self-esteem r = 0.40, (P , .05), DD = 211.0 (P , .005) Change in health r = 0.33 (P , .05), DD = 20.34 (P $ .05) PedsQL child report Severe SCD significantly associated with for 5–18 y increased odds of worse total HRQoL (OR = 4.0 [95% CI: 1.92–8.31]) and psychosocial health domain (OR = 2.81 [95% CI: 1.37–5.77]) by parent proxy but not child report. Severe SCD associated with increased odds of worse physical health domain based on both child and parent reports: parent OR = 5.67 (95% CI: 2.68–11.97), child OR = 3.33 (95% CI: 1.39–7.99). According to parents only, older age (8–12 y and older) is an independent predictor of worse QoL.

CHQ-PF28 CHQ-CF87 Self-report only available for children aged $10 y

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QoL Instrument

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QoL Instrument

10-year-olds only

TACQOL-PF TACQOL-CF

2.7–18.6 CBCL (parent) groups 1–2 (,11 y) Group 3: 11–13 YSR ($11y) No self-report for ,11y Higher scores = greater problems

Age Range (y)

For 11–13 y: Total score, internalizing and externalizing scores by both parent and selfreport were not significantly different between CH children and control subjects. Scores on thought problem (in crossinformant syndrome scales) and somatic problem (in DSM-oriented scales) were significantly higher (P = .012 and P = .024 respectively) in CH patients than controls by parent report but not by self-report. No statistical comparison between child and parent reports given. Children with CH reported lower mean HRQoL on motor, cognitive and social functioning, and on positive emotions (in all P , .0001) than the norm population. Parents of patientswith CH (total group) reported significantly worse HRQoL in their children than parents of the norm population on motor (P , .0001), cognitive (P , .0001), and social (P , .001) functioning and negative emotions (P , .001).


Study Quality

Good Both children and parents reported significantly lower mean HRQoL on motor, cognitive, and social functioning than the norm population. However, in relation to positive and negative emotions the self-reports and parent reports disagreed. Parents overrated scores for positive emotions of their children but underrated scores for negative emotions of their children.

Self-report: scores in all dimensions of Moderate but the behavioral scales were not only n = 16 in significantly different between CH 11–13 y patients and control subjects. group with Parent report: the differences were only self-report in scores on thought problem (in cross-informant syndrome scales) and somatic problem (in DSMoriented scales).

Summary of Findings

CBCL, Child Behavior Checklist; CH, congenital hypothyroidism; CHI, congenital hearing impairment; CHQ-CF87, Child Health Questionnaire–Child Form 87; CHQ-PF28, Child Health Questionnaire–Parent Form 28; CP, cerebral palsy; CP-QoL-Child, Cerebral Palsy Quality of Life Questionnaire for Children; DD, directional difference (mean [SD] = child score – parent score [White-Koning et al11]; mean = parent score – child score [Panepinto et al51]); DSM, Diagnostic and Statistical Manual of Mental Disorders; ICC = intraclass correlation coefficient (ICC values for agreement are considered poor if ,0.40, moderate if 0.40–0.75, and good if .0.75); r, Pearson correlation coefficient; RCT, randomized controlled trial; SCD, sickle cell disease; Spearman, Spearman rank correlation coefficient; TACQOL-CF, TNO-AZL Child Quality of Life Questionnaire Child Form; TACQOL-PF, TNO-AZL Child Quality of Life Questionnaire Patient Form; YSR, Youth Self-Report.

Van der Sluijs Veer et al (2012),58 82 pairs Netherlands 449 controls (children) 465 controls (parents)

Congenital hypothyroidism Bisacchi et al (2011),57 Italy

Author, Location

TABLE 3 Continued

The impact of a condition on selfreported QoL may change during the life course.

The main study strength is the adherence to standard systematic review methods. We used prespecified inclusion criteria and the PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) checklist to guide reporting essential information from the included studies.

Although only 1 reviewer scanned through all abstracts after the initial article selection, only obvious noneligible studies were excluded at this stage. Two second reviewers scanned the titles and abstracts of a 10% sample of the initially identified studies and confirmed decisions on inclusion.

Limitations of this review focusing on self-reported QoL of the younger child group primarily result from the scarcity of such studies and lack of information about the formation of the study sample. For example, many studies, with small sample sizes, reported their results for wide age groups (ie, not separating their results for children and adolescents); this approach greatly reduced the numberof eligible studies forour review. Although we included only studies using validated QoL instruments, there was a wide variability in the instruments used and approaches to statistical analyses, which complicated the synthesis of the findings and resulted in a narrative synthesis only. We assessed the quality of the included studies based on the recruitment criteria, study design, instruments of QoL assessment, and the statistical analyses, which allowed us to build our synthesis on higher quality studies. In studies with low response rates, nonresponse (or selection) bias can be a major concern if nonresponders differ systematically from responders on characteristics that may affect QoL measures. However, the majority of studies did not report the characteristics of responders versus

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nonresponders, thereby impairing study quality. An important objective of the present review was to examine the agreement, or lack of, between children’s and parents’ views on a child’s QoL because this factor is of central importance from both a clinical and research perspective. Unfortunately, there was lack of consistency for the methods used for comparison of self-proxy reports that complicated the synthesis of the findings. In addition, this review only included studies published in English, and we acknowledge that this choice may be another limitation. Determinants of Findings and Comparison With Published Studies Studies assessing children who have different levels of condition severity reported that severity and presence of associated anomalies contributed to the lower scores in some domains of self-reported QoL,33,34,38 which is in agreement with a previous review.1 We observed a lack of consistency in the reviewed studies in relation to QoL in children with congenital conditions, with some reporting comparable36,38,39 or even better40 subjective QoL than control children, whereas others reported lower total or domain-specific scores. This inconsistency cannot be fully attributed to the type of condition, QoL measure used, or age differences, as some studies explored children of the same age groups with the same condition using the same instrument but reported discrepant findings.33,34 The cross-sectional design of the reviewed studies may partially explain the heterogeneity in the findings, because QoL, as a measure of adaptation, is sensitive to context and the impact of the condition, but it is also influenced by other relevant experiences at the time of assessment (eg, change in medication, surgery, schools). These factors can substantially reduce average QoL

scores, which may simply reflect temporary challenges to adaptation for some children. When children with a chronic condition report comparable or better QoL than control subjects, it may indicate that over time the family and the child have adapted to the extra burden of a condition and have developed ways to cope with challenges of everyday life. This adaptation may be particularly true for nonprogressive conditions, such as cerebral palsy, or in children with a correctable congenital anomaly (eg, CHD) but less so for children with a progressive disease (eg, DMD). Studies comparing QoL in case subjects and control subjects according to different age groups found that there were age-related differences; for example, in children with CHD, younger children (5–7 years old) reported better QoL than comparable control subjects, whereas QoL scores for 8- to 12-year-olds were similar.40 Naturally, as values and priorities change during the life course, affected QoL domains vary in different age groups. For example, although family and relationships with parents are more important for younger children, with increasing age, social domains such as “friends” and “perceived support” or “self-esteem” and “school” domains become of growing importance.27,28 Similarly, as physical appearance becomes more important for teenagers, the 8- to 10-year-old children with an orofacial cleft reported a higher QoL average score than the 15- to 18-year-olds, and social-emotional well-being was lower than that in control subjects in this latter age group only.30 As previously reported, health status of the child and the type of condition contributed to self-proxy agreement, but this finding was inconsistent across studies. Thus, using CHQ, discrepancy was reported between self-reports and proxy reports in children with cancer in

50% of 16 items in 4 domains; in the unaffected group, the significant discrepancy was found in 1 item only.12 However, a further systematic review found that child–parent agreement was better for chronically ill children than for healthy children,9 which was not confirmed by a later review reporting self-report–proxy disagreement in both healthy samples (with parents overestimating child QoL) and children with health conditions (with parents underestimating child QoL).61 In children with congenital conditions, there was consistency in studies of children with cerebral palsy, using various measures of QoL; children rated their QoL higher than parents did.11,49 For other conditions, there was less consistency in the direction of discrepancy between self-report and proxy report, with some studies reporting lower scores by parents than children34,45,47,51,56; in other studies, parents reported higher scores than children.31,46,55 Consistent with a previous review,9, we found that QoL domain in studies of children with congenital conditions had a significant impact on self-report–proxy disagreement, with more agreement for objective domains (eg, physical health-related quality of life [HRQoL]) and less for subjective domains (eg, emotional and psychosocial well-being).45,48,51,52,58,59 This finding may be partially explained by parents’ perception of a large effect of functioning level on any dimension of child’s QoL (eg, in children with cerebral palsy), whereas according to self-report, functioning was not associated with emotional and social well-being.52 Similarly, although younger (8–12 years) boys with DMD had significantly higher scores for physical functioning than 13to 17-year-old boys, there was no difference for psychosocial functioning despite the likely disease progression.45 Selfreport–proxy agreement, in particular in subjective QoL domains, also seems to depend on the instrument used; for example, in children with cystic fibrosis, the



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correlation between child and parent scores on emotional domain was much higher with the use of a generic tool (PedsQL) than when using the conditionspecific CFQ.48 Children’s age is also considered a likely factor affecting child–parent agreement. However, only 2 studies included in the earlier review explored the effects of age on parent–child agreement, and the findings were inconclusive.9 In our review, a number of studies reported self-report–proxy agreement across age groups, and the disagreement varied according to age group and QoL domain. For example, for children with cerebral palsy, parents of older children were more likely to proxy report a lower QoL in parental relations than their children, whereas parents of younger children were more likely to proxy report scores lower than children in school domain.11 In children with CHD, there was a significant difference for positive emotional functioning between self-reports of 8- to 11-year-olds and 12to 15-year-olds but not for proxy reports.46 Although child’s pain is an important determinant of self-report– proxy disagreement in children with cerebral palsy,11 parental factors such as parental stress, mental health, and

well-being also contribute to parental underestimation of QoL in children with any condition.9,11 Thus, parental well-being is important to take into account, in particular when no selfreport is available, and also in the interpretation of self-report–proxy disagreement. It is important to assess self-report– proxy agreement, as well as factors affecting it, to distinguish between real differences in a child’s and a parent’s perception of different domains of the child’s QoL and discrepancies resulting from methodologic limitations. Regrettably, in most studies, the level of missing data for specific domains is lacking, and there is no information on young children’s understanding of the questions in the applied questionnaire or on the potential mediating factors (eg, parental mental health) contributing to disagreement. There are also statistical issues in assessment of selfreport–proxy agreement; one-half of the studies did not quantify the level of disagreement, and comparisons in the remainder were based on correlation coefficients only. Correlation coefficients are insufficient for measuring disagreement and agreement, and they should be supplemented by measures

of the direction of disagreement and the rates of agreement.9,11,62 Future studies assessing self-report– proxy agreement in measuring children’s QoL should explore the direction of, and factors contributing to, disagreement; these might include parental well-being, child’s pain, and the use of both generic and conditionspecific QoL tools for comparison.

CONCLUSIONS This review suggests that young children with congenital or early infancy conditions often report scores of QoL comparable to age-matched controls. Their perception ofQoLfrequentlydiffers from that of their parents, in particular for subjective domains. Therefore, even for younger children, both child and parent perspectives are essential to understanding the impact of the condition on a child’s QoL. We did not identify any studies that had assessed QoL repeatedly over time in the same individual, but the sensitivity of such assessments to concurrent events (eg, interventions or medication changes), especially when self-reported, suggests that a longitudinal approach may be useful in clinical practice.

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Self-Reported Quality of Life of Young Children With Conditions From Early Infancy: A Systematic Review Jenni Jardine, Svetlana V. Glinianaia, Helen McConachie, Nicolas D. Embleton and Judith Rankin Pediatrics 2014;134;e1129; originally published online September 22, 2014; DOI: 10.1542/peds.2014-0352 Updated Information & Services

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PEDIATRICS is the official journal of the American Academy of Pediatrics. A monthly publication, it has been published continuously since 1948. PEDIATRICS is owned, published, and trademarked by the American Academy of Pediatrics, 141 Northwest Point Boulevard, Elk Grove Village, Illinois, 60007. Copyright © 2014 by the American Academy of Pediatrics. All rights reserved. Print ISSN: 0031-4005. Online ISSN: 1098-4275.


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