Longitudinal Outpatient and School-Based Service Use among Children with Nonsyndromic Craniosynostosis

Abstract

Background: Although nonsyndromic craniosynostosis has been associated with neurodevelopmental sequelae, a lesser amount of emphasis has been placed on the need for related supportive services. This study assessed the prevalence of such services among children surgically treated for nonsyndromic craniosynostosis and identified predictors of service use. Methods: Parents of children with nonsyndromic craniosynostosis were recruited from an online craniosynostosis support network and surveyed regarding their child’s use of various outpatient and school-based services. Multiple stepwise regression was performed to identify predictive variables for each type of intervention. Results: A total of 100 surveys were completed. Of these, 45 percent of parents reported use of one or more outpatient support services for their children. The most commonly used services were speech therapy (26.0 percent) and physical therapy (22.0 percent), although the use of services such as psychology/psychiatry increased among older children (18.2 percent in children aged 6 to 10 years). Among school-age children (n = 49), the majority of parents (65.3 percent) reported school-based assistance for their children, most commonly for academic (46.9 percent) or behavioral (42.9 percent) difficulties. Significant predictive variables (p < 0.05 following stepwise regression) for increases in various outpatient and school-based services included male sex, African American race/ethnicity, higher parental income, the presence of siblings in the household, increased age at the time of surgery, and sagittal synostosis. Conclusions: Parents of children with nonsyndromic craniosynostosis reported frequent use of outpatient and school-based supportive services throughout childhood. These services may incur a significant burden of care on families. The multifactorial nature of predictive models highlights the importance of cross-disciplinary collaboration to address each child’s longitudinal needs.

Cognitive Outcomes of Children With Sagittal Craniosynostosis Treated With Either Endoscopic or Open Calvarial Vault Surgery

“…Our results suggest that patients in both the endoscopic repair and open repair groups received services at a higher rate than would be expected in the general population (15%) . This finding is similar to that reported by Peck et al As in that earlier study, patients in our study were particularly more likely to receive speech and language therapy than is typically seen in the US. This finding points to a less-studied finding, namely, that patients with a history of craniosynostosis may be more likely than their unaffected peers to require support services well into childhood.…”

Comparison of Emotional and Behavioral Regulation Between Metopic and Sagittal Synostosis

“…Children with repaired NSC are at greater risk for behavioral impairments and require more supportive interventions. [16, 21,22,24,26,29] Suggested etiologies for these neurodevelopmental abnormalities include locoregional compression, increased intracranial pressure, diminished cerebral perfusion, altered intrinsic brain development, and genetic mutations. [6, 30,35] Some studies have found associations between suture type and prevalence and type of behavioral disturbance.…”

“…Additionally, over half of children with metopic synostosis in this cohort were reported to require more supportive services signi cantly higher than the 35% of children with sagittal synostosis. Peck et al has previously described that overall children with NSC require services at a high rate [24]. However, the higher rates of supportive services used in children with metopic synostosis may indicate these children may have more di culties which require more appropriate assistance.Children with sagittal synostosis were found to have more di culties with externalizing behaviors.…”

Discussion: Longitudinal Outpatient and School-Based Service Use among Children with Nonsyndromic Craniosynostosis

“…Throughout his career, John Persing and his team from Yale have attempted to provide answers to questions regarding the neurodevelopmental outcomes in patients with craniosynostosis through a variety of approaches. In this issue, Peck et al 1 report on the use of outpatient and school-based services among children with nonsyndromic craniosynostosis based on a survey of families through the Cranio Kids–Craniosynostosis Support group on Facebook.…”