SEARCH

SEARCH BY CITATION

Abstract

  1. Top of page
  2. Abstract
  3. Method
  4. Results
  5. Discussion
  6. Conclusion
  7. Acknowledgements
  8. References

The literature was systematically reviewed to determine if children with spina bifida have lower self-concept compared with their peers with typical development. Relevant trials were identified by searching electronic databases, supplemented by citation tracking. Of 803 papers initially identified, 15 met the inclusion criteria. Meta-analysis revealed children with spina bifida scored significantly lower than children with typical development for the domains of global self-worth (d=−0.39, 95% confidence interval [CI] −0.65 to −0.12); physical appearance (d=−0.26, 95% CI −0.46 to −0.06); athletic competence (d=−0.45, 95% CI −0.67 to −0.22); social acceptance (d=−0.33, 95% CI −0.55 to −0.11); and scholastic competence (d=−0.43, 95% CI −0.66 to −0.21). There was no difference between the groups for the behavioural conduct domain. Children with spina bifida on average have a lower self-concept than their peers with typical development. Clinicians need to take account of this information in planning the assessment and treatment of this group.

Self-concept is a multidimensional psychological construct that describes what children think of themselves in domains such as social acceptance, athletic competence, scholastic competence, behavioural conduct, and physical appearance. It comprises the child’s perceived identity or their awareness of their own personal characteristics and attributes, and their global sense of self-worth (self-esteem) or how they evaluate their characteristics in relation to others.1 Self-concept is a fundamental component of a child’s psychological health and development.2

Spina bifida is a congenital neural tube defect affecting 1 in every 1000 live births.3 The degree of impairment varies and is dependent on the level and extent of the lesion and the amount of neural tissue involved. Typical impairments include muscle weakness or paralysis, sensory deficits, cognitive deficits, musculoskeletal deformities, and urinary and bowel incontinence,4 all of which may cause difficulties in everyday functional activities such as walking, dressing, and other tasks of personal care. These impairments could conceivably have an impact on the self-concept of children with spina bifida,5 by limiting the child’s ability to explore and interact with others and their environment.6,7 However, there is conflicting evidence on whether children with spina bifida indeed have a lower self-concept.2,5,6,8

Health professionals need to consider the child’s views when deciding optimal management strategies, selecting interventions, and planning for the future.9,10 Understanding the child’s self-concept is an important component of taking account of their views. Recognition of the self-concept of children with spina bifida may also contribute to and enhance the development of the child–clinician relationship. It may help clinicians identify children at risk of lower self-concept, thus facilitating appropriate referral or providing anticipatory guidance.6

To assist clinicians in this regard, a systematic review was conducted to find out if the self-concept of children with spina bifida was different from that of children with typical development. The aims of the review were: (1) to compare the self-concept of children with spina bifida with that of children with typical development for the domains of: global self-worth, physical appearance, athletic competence, social acceptance, behavioural conduct, and scholastic competence; and (2) to investigate if there are differences in self-concept between age, sex, and socioeconomic status.

Method

  1. Top of page
  2. Abstract
  3. Method
  4. Results
  5. Discussion
  6. Conclusion
  7. Acknowledgements
  8. References

Search strategy

Relevant literature was sourced by searching the following electronic databases: AMED (1985–April 2008), CINAHL (1982–April 2008), EMBASE (1988–April 2008), ERIC (1966–April 2008), Medline (1966–April 2008), PEDro (1929–April 2008), PsycINFO (1872–April 2008), PubMed (1966–April 2008) and the Cochrane library. The keywords used were: ‘spina bifida’ or ‘myelomeningocele’ combined with ‘child’ and ‘self-concept’. Relevant synonyms for each term were incorporated into the search strategy (e.g. self-esteem, self-efficacy, and self-perception were used for self-concept). Reference lists of the identified articles were manually searched to identify additional relevant articles. Citation tracking of the included studies and key authors in the area was also conducted using the Web of Science.

Inclusion/exclusion criteria

The following criteria were applied by two independent assessors (NS, YL) to the titles and abstracts of the search yields. Studies were selected if they included children with spina bifida aged under 18 years, compared the self-concept of children with spina bifida with typically developing peers, and measured self-concept using a quantified scale. Studies were excluded if any of the participants had a condition other than spina bifida (e.g. cerebral palsy), focused on outcomes that measured a construct other than self-concept (such as, functional assessment or quality of life), or collected data by qualitative methods (such as unstructured or semi-structured interviews). There was no language restriction. In cases where the title or abstract provided insufficient information about the study, the full text of the article was obtained and read by both assessors. Discrepancies in the decisions made were discussed until a consensus was reached.

Quality assessment

The quality of the studies selected for review was rated using five criteria specific to assessment of observational studies (Table I) adapted from Khan et al.11 These criteria were chosen to ensure the included studies demonstrated an appropriate degree of internal validity and provided the relevant statistics to allow accurate conclusions to be drawn. Each item was scored as either met (two points), partially met (one point) or not met (no points). All articles were assessed independently by two assessors (NT, KD). Both assessors were blinded to the source of the article including the authors and their affiliations, journal name, and publication date. Any disagreements between the two reviewers were resolved by discussion until a consensus was reached.

Table I.   Quality assessment measure of selected studies
NoCriterionSatisfied if:
1Was the study based on a representative sample selected from a relevant population?The report described the source and demographic details (sex, age, level of disability) of the participants with spina bifida and the source for the children with typical development. This item was partially met if only described in detail for children with spina bifida or only described some of the demographics of the children.
2Were the criteria for inclusion and exclusion explicit for both the children with spina bifida and the children with typical development?The report described a list of criteria to determine eligibility for the study for both children with spina bifida and children with typical development. This item was partially met if it only described in detail the criteria for the children with spina bifida.
3Were the two groups comparable on all potential confounding factors?The groups had to be directly comparable for sex and age. The reviewer had to also take into account if socioeconomic status and schooling were comparable. This item was partially met if age and sex were comparable but other factors might have been confounding.
4Did the outcome measurement tools used demonstrate sufficient validity for comparing the self-concept of the groups?This item was met if the self-concept measurement tool was referenced and either stated that the tool has demonstrated evidence of validity with a reference, or reports that the tool had been able to detect changes or differences in a relevant population (e.g. children with physical disabilities) with a reference.
5Was an appropriate statistical analysis used?This item was partially met if only means and SDs were reported. This item was partially met if it is just reported that the tool has good measurement properties or reliability, without mention of validity. The report provided means and SDs (or medians and interquartile range) for each group for the measure of self-concept, and reported an appropriate statistical test for comparison (t-test or equivalent non-parametric test such as Mann-Whitney U).

Data extraction and statistical analysis

Data were extracted from the included studies by two independent reviewers (NT, KD) using a standardized data extraction form developed for the review. Details of the study objective, study design, participant characteristics (including age, sex, and background), outcome measures used, main results, and study limitations were documented.

Standardized mean differences (effect sizes d) were calculated by subtracting the mean of the children with typical development from the mean of the group with spina bifida, and dividing by the pooled standard deviation (SD). Accordingly, the included studies needed to report the means and SDs for both groups of children. Calculations were completed using web-based software.12 Standardized mean differences less than 0.20 were considered small, between 0.20 and 0.50 were considered medium, and greater than 0.80 were considered large.13

Meta-analysis was conducted to provide an overall estimate of the effect for each of the domains where outcomes were considered similar enough to pool. To account for any heterogeneity in the data, a random effects model was applied, as this model assumes variability due to a combination of random sampling error and systematic sources of variation. MetaView software (version 5.3)14 was used for all calculations. To investigate if the review was subject to publication bias, funnel plots were prepared by plotting the inverse of the standard error of the effect size estimate against the effect size.15 These were examined for asymmetry. Where a study was found to report disparate results, sensitivity analysis was conducted to investigate the impact of that study on the findings.

Results

  1. Top of page
  2. Abstract
  3. Method
  4. Results
  5. Discussion
  6. Conclusion
  7. Acknowledgements
  8. References

As shown in Figure 1, of the 803 papers located by the search strategy, 15 were included in the review.5–9,16–25 These studies included 1340 participants ranging in age from 4 years 1 month to 18 years 11 months, and a relatively equal distribution of males and females (Table II). The majority of children with spina bifida had a lesion located in the lumbar or sacral region, used a device to assist with their mobility (e.g. a brace or wheelchair), and attended a mainstream school. However, children with spina bifida with a range of lesions and mobility levels were represented. Of the six studies9,16,18,19,24,25 that reported the type of school attended by the participants with spina bifida, only four9,18,19,24 stated the children were taught in a regular classroom; the other two studies16,25 did not specify the type of classroom the participants were in. Seven studies8,9,17,18,21,24,25 recorded the presence or absence of hydrocephalus or a shunt but none explored the effect of these variables on self-concept. The comparison group participants of typically developing peers were mostly matched for age and sex.

image

Figure 1. Study selection process.

Download figure to PowerPoint

Table II.   Summary of included studies
AuthorQA /10Sample sizeAgeSexIQSchoolingLevel of LesionHydrocephalus/ shunt statusMobilityComparison groupOutcome measure
  1. QA; quality assessment; SB, spina bifida; C, comparisons; M, males; F. females; PPVT-R, Peabody Picture Vocabulary Test – Revised; T/sp, thoracic spine; L/sp, lumbar spine; MMC, myelomeningocele; LMC, lipomeningocele.

Appleton et al.96158 79 SB, 79 C13y 7mo SB 13y 3mo C (7-18y 11mo)76M 82 F 38M 41F in each group78.9 (SD7.9) SB 100.8 (SD 14.9) C49 SB mainstream, 17 special school, 13 other 63 C mainstreamLumbar 26 Sacral 11 Thoracic 30 Cervical 370.9% had hydrocephalus 65.8% had surgery for shuntNo aids 16 Braces 31 Wheel chairs or carried 23Matched for age (SD 6mo), sex, classroom & housing neighbourhood. No known chronic illness, disability or special educational needsSelf-perception Profile for Learning Disabled students (Renick & Harter29)
Appleton et al.187144 72 SB 72 C14y SB (7-18y 11mo)35M 37F78.6 (SD 17.9) SB45 mainstream, 14 special school, 13 other (SB)Not reported70.9% had hydrocephalus 65.8% had surgery for shuntNot reportedMatched for age (SD 6mo), sex, classroom & housing neighbourhood. No known chronic illness, disability or special educational needsSelf-perception Profile for Learning Disabled students (Renick & Harter29)
Campbell et al.7540 20 SB 20 C14y 4mo (SD 2y 5mo) SB 14y 3mo (SD 2y 5mo) C8M and 12 F in each group99.05 (SD 9.61) SBNot reportedLumbar 16 Sacral 4Not reportedNot reportedMatched for age and sexTennessee Self-concept scale (Fitts30)
Casari & Fantino226248 40 SB 208 C12y (SD 8–18y) SB Control group not stated20M 20F SB 106M 102F C70-120 (mean 90) for SB Controls not statedNot reportedNot reportedNot reportedNot reportedNot reportedPerceived competence scale for children (Harter26)
Edwards-Beckett20660 30 SB 30 C9y 6mo (SD 2y) SB C 9y 6mo (SD 1y 8mo)12M 18F SB 15M 15F CChildren were in an appropriate school grade for their ageNot reportedLumbo-sacral 52% Lumbar 19% Thoraco-lumbar 19% Sacral 10%Not reportedNot reportedNot reportedPiers-Harris Self-Concept Scale for children (Piers31)
Ellis251062 31 SB 31 C15.3 (13–17y 6mo) Ages of control group not stated13M 18F in each group>70All attended mainstream schools (30 public, 1 private)Lumbar 20 Sacral 1067.7% had hydrocephalus No information on shunt statusNo aids (14/31) Braces/ crutches (8/31) Wheelchair (9/31)Matched for age, sex, ethnicity, socioeconomic status & type of school attendedPiers-Harris Self-Concept Scale for children (Piers31) Tennessee Self-concept Scale (Fitts30)
Harvey & Greenway16627 9 SB 18 CMean 10y 3mo for C group Not stated for SB group8M 10F C Not stated for SB groupIntelligence within normal range5 mainstream, 4 special school SB 18 mainstream C6 myelomen-ingoceles 3 meningocelesNot reportedNot reportedRecruitment through guiding and scouting movements. Children were selected by leaders of these organizationsPiers-Harris Self-Concept Scale for children (Piers31)
Holmbeck et al.86126 59 SB 67 C8y 4mo (SD 6mo) SB 8y 6mo (SD 6mo) C (8–9y)37M 31F in each groupPPVT-R scores 92.49 (SD 18.49) SB 108.97 (SD 15.06) CNot reportedT/sp 13% L/sp 54% Sacral 32% 82% MMC 12% LMC 6% otherNo information on hydrocephalus 71% had a shunt in situMobile with no assistance 19% With braces 63% By wheelchair 18%Recruited by contacting school where children with SB were enrolledSelf-perception Profile for Children (Harter26)
Kazak & Clark23662 30 SB 32 CNot stated but < 18yNot statedNot reportedNot reportedNot reportedNot reportedNot reportedRecruited through local paediatricians, general paediatric clinics and a newspaper advertisementPiers-Harris Self-Concept Scale for children (Piers31)
Landry et al.17730 15 SB 15 C8y 2mo SB 8y 1mo C9M 6F SB 6M 9F CIntelligence within normal rangeNot reportedNot reported100% had hydrocephalus 100% had a shunt in situ8 were ambulatory and 7 were non-ambulatoryRecruited from enrolment list of two community day care centresPictorial scale of perceived competence & acceptance for young children (Harter & Pike32) Self-perception Profile for Children (Harter26)
MacBriar24449 19 SB 30 C12y (SD2.6) 5y SB 11.5 (SD 2). 13y C9M 10F (SB) 11M 19F C1 child diagnosed with intellectual disability Data not presented for 1 childAll children with two exceptions attended mainstream schoolsLumbar 3 Sacral 1 Don’t know 1557.9% had hydrocephalus 57.9% had a shunt in situBraces 12 (6 long leg, 6 short leg) Ambulatory 4 Non ambulatory 2Siblings of the SB group were used as controlsPiers-Harris Self-Concept Scale for children (Piers31)
Mobley et al.6642 21 SB 21 C64.7mo SB 64.3mo C7M 14F in each groupNot reportedNot reportedAll MMCNot reportedAll used mobility assistive devices with 67% using more than 1 deviceRecruited through Spina Bifida Association (siblings of children with SB), day care centres and by personal contacts in the communityPictorial Scale of Perceived Competence and Social Acceptance for Young Children (Harter & Pike32)
Pearson et al.51122 80SB 42C‘12 yr olds’Not statedNot statedNot reportedNot reportedNot reportedNot reportedNot reportedPiers-Harris Self-Concept Scale for children (Piers31)
Rodriguez Aponte19940 20 SB 20 CMean age 10y 1mo (9–12y 5mo)10M 10F in each groupNot stated but children matched for grade and grade point averageAll attended mainstream schoolsModerate level of MMCNot reportedAll mobile with aid, 40% minimal usage, 60% use aid regularlyThree public schools in Puerto Rico Matched for age, sex, grade, socioeconomic status and grade point averagePiers-Harris Self-Concept Scale for children (Piers31)
Thill et al.217130 62 SB 68 C 8y 4mo (SD 6mo) SB 8y 6mo (SD 6mo)37M 31F in each groupNot statedNot reportedT/sp 13% L/sp 54% Sacral 32% 82% MMC 12% LMC 6% otherNo information on hydrocephalus 71% had a shunt in situMobile with no assistance 18% With braces 63% By wheelchair 19%Recruited by contacting school where children with SB were enrolledSelf-perception Profile for Children (Harter26)

All studies examined self-concept as a multidimensional construct using a variety of self-concept measures (Table III). Six studies, however, only reported scores for the domain of global self-worth despite using a multidimensional scale.5,18–20,22,25 Where contact details were provided, authors of these studies were contacted and supplementary information not included in their published manuscript was requested to assist with data analysis. Of the eight authors contacted, three responded, however, additional information was only available from one author (G Holmbeck).

Table III.   Outcomes measures used to assess self-concept
ScaleNo of itemsDescriptionRespondentExampleStudies
  1. *Representative sample items from the TSCS:2 Adult Form copyright © 1996 by Western Psychological Services. Reprinted by N. Shields, LaTrobe University, for the sole purpose of scholarly review. Not to be reprinted in whole or in part for any other purpose without the prior, written authorization of WPS, 12031 Wilshire Boulevard, Los Angeles, CA 90025.

Piers-Harris Self-concept Scale for children3180Statements to which the child answers yes or noChild‘I am well behaved at school’Edwards-Beckett20; Ellis25; Harvey & Greenway16; Kazak and Clark23; MacBriar24; Pearson et al.5; Rodriguez19
Self-perception Profile for Children2636Each question presents two conflicting statements. One that most reflects the child is chosen and subsequently the child decides if it is sort of true or really true for themChildSome kids find it hard to make friends BUT other kids find it’s pretty easy to make friendsCasari and Fantino22; Landry et al.17; Thill et al.21; Holmbeck et al.8
Self-perception Profile for learning disabled students2946Each question presents two conflicting statements. One that most reflects the child is chosen and subsequently the child decides if it is sort of true or really true for themChildSome kids read pretty fast BUT other kids are pretty slow readersAppleton et al.9; Appleton et al.18
Pictorial Scale of Perceived Competence and Acceptance for young children3224Child looks at two pictures and is asked which of the children in the two pictures is most like him or herChildQuestions are in pictorial formatLandry et al.17; Mobley et al.6
Tennessee Self-concept Scale30100Statements with five possible responses: completely false, mostly false, partly false- partly true, mostly true, completely trueChild‘I am a friendly person’*Campbell et al.7; Ellis25

The median quality assessment score was 6 out of 10 (range 1−10). Only four out of the 15 included studies specified their criteria for inclusion and exclusion for both children with spina bifida and typically developing peers (item 2). The two groups were comparable for all potential confounding factors in only five studies.

Global self-worth

Global self-worth is an overall judgement about how a child perceives their worth or value as a person (i.e. their self-esteem) including the extent to which they like themselves and are happy with the way they are leading their life.25 Twelve studies compared the global self-worth of children with spina bifida with their typically developing peers. Ten studies concluded there was no difference between the groups for global self-worth (Fig. 2), while two studies found global self-worth was lower in children with spina bifida.5,16 Meta-analysis revealed that global self-worth of children with spina bifida was significantly lower than children with typical development with a medium-sized effect (d=−0.39, 95% CI −0.65 to −0.12; Fig. 2).

image

Figure 2. Standardized mean differences and 95% confidence intervals for global self-worth.

Download figure to PowerPoint

Physical appearance

The physical appearance domain explores how children feel about the way they look.26 Data for the physical appearance domain of self-concept were reported in seven studies (Fig. 3). Five studies found no difference between children with spina bifida and their typically developing peers,9,16,17,23,24 while two studies reported children with spina bifida scored lower on this domain than their typically developing peers. Meta-analysis revealed children with spina bifida scored significantly lower than children with typical development on the domain of physical appearance with a medium-sized effect (d=−0.26, 95% CI −0.46 to −0.06; Fig. 3).

image

Figure 3. Standardized mean differences and 95% confidence intervals for physical appearance, athletic competence, social acceptance and scholastic competence.

Download figure to PowerPoint

Athletic competence

The athletic competence domain explores a child’s perceived competence in their ability to participate in sports.26 Four studies assessed the domain of athletic competence; all used a version of the Self-Perception Profile for Children.6,9,17,21 Meta-analysis including three of these studies (Mobley et al.6 did not report standard SDs for their cohort and so their data could not be included) found children with spina bifida scored lower than their peers with typical development on this domain with a medium-sized effect (d=−0.45, 95% CI −0.67 to −0.22; Fig. 3).

Social acceptance

The social acceptance domain evaluates how a child feels about how they get along with their peers.26 Four studies also assessed the domain of social acceptance using the Self-Perception Profile for Children.6,9,17,21 Three studies concluded there was no difference between the groups for social acceptance (Fig. 3), while one study found social acceptance was lower in children with spina bifida.9 Data from three of these studies were included in a meta-analysis (as Mobley et al.6 did not report SDs for their cohort, their data could not be included) and results found scores for children with spina bifida overall were significantly lower for this domain compared with their peers with typical development with a medium effect size (d=0.33, 095% CI −0.55 to −0.11; Fig. 3).

Three studies16,23,24 used the Piers-Harris questionnaire31 which includes a domain related to social acceptance (popularity), however, overall there was no difference between the groups when a meta-analysis was conducted (d=−1.10, 95% CI −2.62 to 0.41; Fig. 3).

Scholastic competence

The scholastic competence domain evaluates how children feel about how they perform at school.26 Four studies examined the domain of scholastic competence using a version of the Self-Perception Profile for Children.26 Three of these studies were included in a meta-analysis which found children with spina bifida had a lower self-concept than their typically developing peers with a medium-sized effect (d=−0.43, 95% CI −0.66 to −0.21; Fig. 3). The fourth study6 also reported a significant difference between the groups (t=−2.8, p=0.008). Additionally, three studies assessed the related domain of intellectual competence and school status using the Piers-Harris Scale, which evaluates a child’s competence in schoolwork but also includes items relating to behaviour, anxiety, physical attributes, and popularity. These studies found no difference between children with spina bifida and children with typical development (Fig. 3).

Behavioural conduct

The behavioural conduct domain explores a child’s perceived competence in how they behave.26 There was no difference between the scores of children with spina bifida and their peers with typical development for any of the behavioural conduct domains (Fig. 4).

image

Figure 4. Standardized mean differences and 95% confidence intervals for behavioural conduct.

Download figure to PowerPoint

Effect of age, sex, and socioeconomic status on self-concept

There were insufficient data to perform meta-analysis on subgroups of participants according to age, sex, and socioeconomic status (as rated by Hollingshead Four Factor Index; Hollingshead AA, 1975, unpublished material). Two studies9,18 found a main effect for sex on self-concept but another study24 did not. Three studies9,19,24 found no effect of age on self-concept, although another study22 reported adolescents with spina bifida (aged 12−17y) scored lower on global self-worth than pre-adolescents with spina bifida (aged 8−11y; p<0.01). No difference was found between children with spina bifida and their typically developing peers from similar socioeconomic backgrounds for global self-worth, behavioural conduct, and social acceptance.5,8 Children with spina bifida, however, scored lower on athletic competence and scholastic competence irrespective of socioeconomic background.8

Sensitivity analysis

Examination of the funnel plots found one study16 appeared to have disparate results from the other included studies (Fig. 5). Sensitivity analyses were conducted to explore if removing the data from this study from the meta-analysis would affect the findings of the review. Excluding this trial did not change the results in the domains of global self-worth (d=−0.31, 95% CI −0.45 to −0.19); physical appearance (d=−0.24, 95% CI −0.49 to −0.002); behavioural conduct (d=−0.18, 95% CI −0.4 to 0.05); intellectual (d=−0.24, 95% CI −1.03 to 0.54; and happiness (d=−0.35, 95% CI −0.73 to 0.03). However, an effect for the popularity (d=−0.41, 95% CI −0.8 to −0.03) and anxiety d=−0.7, 95% CI −1.09 to −0.31) domains of the Piers-Harris scale was found when this trial was removed from the analysis. It is possible that these differences reflect the small sample size (n=9) of children with spina bifida in this study. It is also probable that selection bias was an issue in this study as the children from the comparison group with typical development were recruited through the guiding and scouting movements and were selected by leaders of these organizations to participate in the study.

image

Figure 5. Funnel plot to investigate for publication bias.

Download figure to PowerPoint

Discussion

  1. Top of page
  2. Abstract
  3. Method
  4. Results
  5. Discussion
  6. Conclusion
  7. Acknowledgements
  8. References

Meta-analysis revealed medium-sized (d=−0.26 to d=−0.45) and significant standardized mean differences to the effect that children with spina bifida scored significantly lower than their peers with typical development in the domains of global self-worth, physical appearance, athletic competence, social acceptance, and scholastic competence. It might not be unexpected for children with spina bifida to have a lower self-concept given that many of the characteristic symptoms associated with spina bifida such as incontinence, muscle paralysis, and musculoskeletal deformities may impact on their ability to function in their environment and interact with others. Indeed, children with spina bifida have been shown to be more dependent on adults, less likely to make independent decisions, have fewer social contacts outside of school, have lower scholastic abilities, and be less active than their typically developing peers.8 Children with spina bifida often demonstrate poorer motor skills which can affect their play17 and their ability to complete their activities of personal care.18 This regular reminder of their physical disability and limitations might cue more frequent negative perceptions of their selves, resulting in frustration at their poorer motor skills and in less personal satisfaction in carrying out and completing tasks. These findings also show that they are aware they are different from their peers with typical development9 and know that it is harder for them to take part in games and sports (athletic competence). Their lower self-concept in the social acceptance domain is consistent with previous literature that suggests children with disabilities often feel isolated from their peers.9,27

Although 10 of the 12 included studies reported no difference in the global self-worth of children with spina bifida and their peers with typical development, meta-analysis found a small but significant effect (d=−0.39) for this domain. Based on this effect size, a study would need to include at least 103 participants in each group to detect any difference in global self-worth between the groups. As none of the studies had included this number of participants, it would appear their sample sizes were too small to detect any overall difference. These results demonstrate the value of meta-analysis; the evidence from individual studies when viewed in isolation gave the impression of no difference; however, when the trends observed in these studies were aggregated, a significant difference was found.

The majority of participants with spina bifida attended mainstream schools, and are likely to have compared themselves to their typically developing peers rather than other children with spina bifida, which may reinforce the concept that they were different.16,18 Children with spina bifida who attend mainstream schools place more importance on physical appearance, use social comparison processes to evaluate their own physical appearance, and feel less attractive when comparing themselves with their peers with typical development than to other children with physical disabilities.9 These children are in a difficult position; they identify with their peers with typical development but may feel less competent and less accepted than those to whom they compare themselves.9

These results have implications for health professionals working with children with spina bifida who assist in the decision making processes about the management of a child’s disabilities. Clinicians need to be aware that on average, children with spina bifida will have a lower self-concept; routine, and ongoing periodic measurement of self-concept in clinical practice, using an outcome measure with sound psychometric properties, such as the Self-Perception profile for children, would assist clinicians in being informed about how children with spina bifida feel about themselves. Such information would help clinicians identify children with spina bifida who have a lower self-concept, allowing them to work in partnership with the child to develop coping strategies.9,24 Clinicians might help the child manage their negative body image through appropriate referral for cognitive and behavioural self-management interventions.18 They might help negotiate child independence and autonomy,18 e.g. helping them master those physical and cognitive skills they are capable of6 and assist the child to discover development opportunities where they feel competent,18 e.g. meeting other children with spina bifida or other physical disabilities perhaps through specialized camps or involvement in wheelchair sports. Such experiences may be valuable in helping children feel ‘part of a peer group’ or a larger community.9

It is important to remember, however, that the findings do not indicate that all children with spina bifida will have a low self-concept, only that this is the case on average. While it is useful for health professionals to know how an average group of children with spina bifida feel about themselves, in general they work with individual children and so cannot assume every child will have a low self-concept. Individual assessment, therefore, is very important when dealing with children who are unhappy with themselves, as this may not necessarily relate to their impairment, and so clinicians must be open to looking beyond the disability itself.

The strengths of the review are that it used an extensive search strategy to locate 15 studies including 1340 participants and advanced the literature in this area by conducting a robust meta-analysis. Many of the included studies employed the same or similar outcome measures (e.g. Self-perception Profile for children and the Piers–Harris Self-concept Scale for children), making the results from the meta-analysis more robust. In a majority of cases the findings of these studies were consistent with each other. This review was limited in determining the effect of demographic characteristics such as age, sex, and socioeconomic status on self-concept in children with spina bifida because with only one exception, all the included trials did not report data for individual subgroups and so meta-analysis was not performed due to a lack of data. A further limitation is that although the random effects model assumed variability was due to a combination of random sampling error and systematic sources of variation, heterogeneity may still be an issue. Meta-analysis also cannot compensate for limitations in the original data such as the presence of uncontrolled confounding variables, measurement error, and selection bias.28

Conclusion

  1. Top of page
  2. Abstract
  3. Method
  4. Results
  5. Discussion
  6. Conclusion
  7. Acknowledgements
  8. References

Children with spina bifida on average have a lower self-concept than their peers with typical development. Clinicians need to take account of this information in planning the assessment and treatment of this group. Finally, this review demonstrated the benefit of using meta-analysis to detect a moderate effect by improving the power of a number of small studies.

Acknowledgements

  1. Top of page
  2. Abstract
  3. Method
  4. Results
  5. Discussion
  6. Conclusion
  7. Acknowledgements
  8. References

We would like to thank Yijun Loy (YL) for her contribution as an independent reviewer in assessing the titles and abstracts of the database searches. We would also like to thank Kylee Doust for her assistance in translating the Casari and Fantino22 manuscript from Italian into English.

References

  1. Top of page
  2. Abstract
  3. Method
  4. Results
  5. Discussion
  6. Conclusion
  7. Acknowledgements
  8. References
  • 1
    Gallahue D, Ozmun J. Understanding motor development: infants, children, adolescents, adults. Boston: McGraw Hill, 1998.
  • 2
    Miyahara M, Piek J. Self-esteem of children and adolescents with physical disabilities: quantitative evidence from meta-analysis. J Dev Phys Dis 2006; 18: 21934.
  • 3
    McLone DG, Ito J. An introduction to spina bifida. Chicago, IL: Children’s Memorial Spina Bifida Team, 1998.
  • 4
    Campbell S, Vander Linden D, Palisano R. Physical therapy for children. 3rd edn. St Louis: Saunders Elsevier, 2006.
  • 5
    Pearson A, Carr J, Halliwell M. The self concept of adolescents with spina bifida. Z Kinderchir 1985; 1: 2730.
  • 6
    Mobley CE, Harless LS, Miller KL. Self-perceptions of preschool children with spina bifida. J Pediatr Nurs 1996; 11: 21724.
  • 7
    Campbell M, Hayden P, Davenport S. Psychological adjustments of adolescents with myelodysplasia. J Youth Adolesc 1977; 6: 397407.
  • 8
    Holmbeck GN, Westhoven VC, Phillips WS, et al. A multimethod, multi-informant, and multidimensional perspective on psychosocial adjustment in preadolescents with spina bifida. J Consult Clin Psychol 2003; 71: 78296.
  • 9
    Appleton PL, Minchom PE, Ellis NC, Elliott CE, Boll V, Jones P. The self-concept of young people with spina bifida: a population-based study. Dev Med Child Neurol 1994; 36: 198215.
  • 10
    Thomas A, Bax M, Smyth D. The health and social needs of young adults with physical disabilities. Clinics in Developmental Medicine No. 106. London: Mac Keith Press, 1989.
  • 11
    Khan K, Riet G, Popay J, Nixon J, Kleijnen J. Phase 5: Study quality assessment. In: Undertaking systematic reviews of research on effectiveness CRD’s guidance for those carrying out or commissioning reviews, 2001. http://www.york.ac.uk/inst/crd/report4.htm (accessed 12 May 2008).
  • 12
    Curriculum Evaluation and Management Centre. 2007 ( http://www.cemcentre.org) (accessed 4th July 2008).
  • 13
    Cohen J. Statistical power analysis for behavioural sciences. New York: Academic Press, 1977.
  • 14
    Schwarzer R. Meta-Analysis Programs. Version 5.3. Berlin: Freie Universität, 1995.
  • 15
    Higgins JPT, Green S, editors. Cochrane handbook for systematic reviews of interventions 4.2.6, 2006. http://www.cochrane.org/resources/handbook/hbook.htm (accessed 12th May 2008).
  • 16
    Harvey DH, Greenway AP. The self-concept of physically handicapped children and their non-handicapped siblings: an empirical investigation. J Child Psychol Psychiatry 1984; 25: 27384.
  • 17
    Landry SH, Robinson SS, Copeland D, Garner PW. Goal-directed behavior and perception of self-competence in children with spina bifida. J Pediatr Psychol 1993; 18: 38996.
  • 18
    Appleton PL, Ellis NC, Minchom PE, Lawson V, Boell V, Jones P. Depressive symptoms and self-concept in young people with spina bifida. J Pediatr Psychol 1997; 22: 70722.
  • 19
    Rodriguez Aponte M. A study of the self-concept of handicapped and nonhandicapped children and the parental perceptions of their offsprings’ self-concept: A comparative analysis. (Thesis). Temple University, 1989.
  • 20
    Edwards-Beckett J. Parental expectations and child’s self-concept in spina bifida. Child Health Care 1995; 24: 25767.
  • 21
    Thill ADW, Holmbeck GN, Bryant FB, Nelson C, Skocic A, Uli N. Assessing the factorial invariance of Harter’s self-concept measures: comparing preadolescents with and without spina bifida using child, parent, and teacher report. J Pers Assess 2003; 81: 11122.
  • 22
    Casari EF, Fantino AG. Spina bifida adolescents self-esteem development. Ricerche di Psicologia 1996; 20: 22938.
  • 23
    Kazak AE, Clark MW. Stress in families of children with myelomeningocele. Dev Med Child Neurol 1986; 28: 22028.
  • 24
    MacBriar BR. Self-concept of preadolescent and adolescent children with a meningomyelocele. Issues Compr Pediatr Nurs 1983; 6: 111.
  • 25
    Ellis CS. Adolescents with chronic illness: spina bifida and the perception of self. (Thesis). Fielding Inst., 1994.
  • 26
    Harter S. Manual for the self-perception profile for children. Denver, CO: University of Denver, 1985.
  • 27
    Lord J, Varzos N, Behrman B, Wicks J, Wicks D. Implications of mainstream classrooms for adolescents with spina bifida. Dev Med Child Neurol 1990; 32: 2029.
  • 28
    Greenland S. Can meta-analysis be salvaged? Am J Epidemiol 1994; 140: 78387.
  • 29
    Renick MJ, Harter S. Impact of social comparisons on the developing self-perceptions of learning disabled students. J Educ Psychol 1989; 81: 63138.
  • 30
    Fitts W. Manual for Tennessee Self-Concept Scale. Nashville, TN: Counselor Recordings and Tests, 1965.
  • 31
    Piers E. Manual for the Piers-Harris Children’s Self-concept Scale. Nashville, TN: Counsellor Recordings and Tests, 1969.
  • 32
    Harter S, Pike R. The pictorial scale of perceived competence and social acceptance for young children. Child Dev 1984; 55: 196982.