Psychosocial Adjustment in Children and Adolescents with a Craniofacial Anomaly: Diagnosis-Specific Patterns

The Cleft Palate Craniofacial Journal

Speltz

et al. 2013

Objective To evaluate the hypothesis that children with craniosynostosis and their parents have differences in psychosocial outcomes, as compared with an unaffected control group. Design Two studies were conducted, both which followed children born with and without craniosynostosis. Study 1 ascertained affected children from clinics and Study 2 ascertained affected children from a population-based study of birth defects. Participants Study 1 included 22 children with single-suture craniosynostosis and 18 controls, ages 4–5 years. Study 2 included 24 children with non-syndromic craniosynostosis and 124 unaffected controls, ages 5–9 years. Main Outcome Measures Outcome measures included the Child Behavior Checklist (CBCL), Social Competence Scale (SCS), Pediatric Quality of Life Inventory (PedsQL™), and Parenting Stress Index (PSI). Results We observed lower scores on measures of health-related quality of life in cases versus controls, with adjusted effect sizes ranging from −0.72 to −0.44 (p<0.05) on summary measures. Small but statistically non-significant increases in behavioral problems were observed in cases versus controls, with no apparent differences in social competence or parenting stress. Conclusions Results provide preliminary evidence suggesting that children with non-syndromic craniosynostosis may have elevated risk for psychosocial difficulties, particularly health-related quality of life. Continued follow-up through pre-adolescence and adolescence is warranted.

Section: Discussionmentioning

confidence: 99%

Psychosocial Outcomes in Children with and without Non-Syndromic Craniosynostosis: Findings from Two Studies

Cloonan

The Cleft Palate Craniofacial Journal

Speltz

et al. 2013

“…12 However, most existing studies have been limited by small sample sizes of children with HFM (fewer than 100 cases) relative to the complex and highly variable HFM phenotype, ranging from isolated microtia to severe bilateral facial malformations. 12,13 Dufton et al 14 also noted that the broad age ranges and lack of demographically matched control groups in previous studies limited the generalizability of results to a broader population of children with HFM and their families. To our knowledge, there is no large-scale study examining the HRQOL for children with HFM.…”

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confidence: 99%

Health-Related Quality of Life in Children with Hemifacial Microsomia

Khetani

Journal of Developmental &Amp; Behavioral Pediatrics

Speltz

et al. 2013

Purpose To compare health-related quality of life (HRQOL) among children with and without hemifacial microsomia (HFM) as assessed by parents and the children themselves during the elementary school years. Methods One hundred thirty-six children with HFM (49 females, mean age = 6 years, 11.9 months, SD = 1.004) were compared with 568 matched controls (285 females, mean age = 6 years, 10.2 months, SD = 0.998) for parent and child responses on the PedsQL Version 4.0. Results After adjustment for sociodemographic factors, parent-reported summary scores were worse for affected children than control group children for physical (effect sizes [ES] = 0.26, p = .004), social (ES = 0.34, p = .001), and school (ES = 0.32, p = .001) functioning. There were no significant mean differences in summary scores based on children’s self-reported functioning. Conclusions Case-control mean differences in HRQOL were more apparent based on parent report, but not child self-report. Summary score findings suggest that case parents have concerns about their child’s HRQOL, particularly with respect to their child’s physical, social, and school functioning. Additionally, our findings highlight the potential differences between child and parent perspectives and the importance of collecting data from multiple reporters.

“…In two of these studies, the Child Behavior Checklist (CBCL) was given to children with SSC in hospital-based craniofacial programs. 5, 6 The third study relied on parent report to assess behavioral problems among 63 children with metopic craniosynostosis evaluated over a 10 year period. 7 The fourth study featured a population-based cohort in which mothers of 82 children with SSC were interviewed by telephone.…”

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confidence: 99%

Behavioral Adjustment of Toddler and Preschool-Aged Children with Single-Suture Craniosynostosis

Kapp-Simon

Plastic &Amp; Reconstructive Surgery

Barr-Schinzel

et al. 2012

Background The purpose of this study was to confirm initial reports of elevated behavior problems in children with single-suture craniosynostosis (SSC), using multiple informants, longitudinal analyses and a control group. We hypothesized higher levels of maladjustment for children with SSC than comparison children, particularly at the older age and in selected areas of previously observed vulnerability: attention and social adjustment. Method A Child Behavior Checklist (CBCL) was completed when children were ~19 months by 436 mothers (219 with SSC) and 371 fathers (177 with SSC); and at ~37 months by 361 mothers (175 with SSC) and 303 fathers (142 with SSC). A minimum of one caregiver/teacher report was available for 169 of these children (74 with SSC) using the Caregiver-Teacher Report Form (CTRF). Results Average CBCL/CTRF externalizing, internalizing and total scores for all informants were consistently higher (worse) for children with SSC than control group children, but most differences were small and statistically non-significant. No differences associated with suture site were found. At the oldest age point, both mothers and fathers (but not teachers) generated higher average scores for patients than for controls on scales measuring attention and social problems, with small to medium effects sizes (0.20 to 0.32). Conclusion On average toddlers/preschoolers with SSC show behavioral development that is largely indistinguishable from same-aged peers of similar socioeconomic background. The predictive significance of small group differences in attention and social adjustment will be assessed in a follow-up of this cohort at age 7.