Dataset Information

Visuomotor Function in School-Age Children with Single-Suture Craniosynostosis.

ABSTRACT: Previous studies have shown that infants and young children with single-suture craniosynostosis (SSC) perform more poorly on tests of visuomotor function than children without SSC. However, previous studies are limited by small sample sizes and little is known about the persistence of visuomotor problems into the school-age years. The aim of this study was to compare visuomotor function in children with and without SSC at the beginning of elementary school.The study included 179 children with SSC (cases) and 183 children without SSC (controls). Visuomotor function was measured by the NEPSY-II Arrows, the Purdue Pegboard Test, and the Beery-Buktenica Developmental Test of visual-motor integration. Case-control differences were estimated using linear regression, adjusted for age, sex, socioeconomic status, and maternal IQ.Cases scored more poorly on all measures of visuomotor function, although the magnitude of case-control differences varied across measures. The greatest differences were observed for the Purdue Pegboard Test, with an average adjusted difference of -0.2 to -0.4 SD points (p-values ranged from .008 to .05). Case-control differences were small in magnitude for other measures of visuomotor function, ranging from -0.01 to -0.1 SD points (p-values ranged from .22 to .88).Children with SSC experienced deficits in manual dexterity into the school-age years but were similar to children without SSC on measures of visual processing. These findings advocate for the assessment of fine-motor function as part of school readiness evaluations in children with SSC.

SUBMITTER: Wallace ER

PROVIDER: S-EPMC4930420 | biostudies-other | 2016 Jul-Aug

REPOSITORIES: biostudies-other

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Visuomotor Function in School-Age Children with Single-Suture Craniosynostosis.

Wallace Erin R ER Collett Brent R BR Kapp-Simon Kathleen K Starr Jacqueline R JR Birgfeld Craig C Speltz Matthew L ML

Journal of developmental and behavioral pediatrics : JDBP 20160701 6

<h4>Objective</h4>Previous studies have shown that infants and young children with single-suture craniosynostosis (SSC) perform more poorly on tests of visuomotor function than children without SSC. However, previous studies are limited by small sample sizes and little is known about the persistence of visuomotor problems into the school-age years. The aim of this study was to compare visuomotor function in children with and without SSC at the beginning of elementary school.<h4>Methods</h4>The s ...[more]

PMID: 27355879

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Project description:BACKGROUND: Craniosynostosis, the premature fusion of calvarial sutures, is a common craniofacial abnormality. Causative mutations in more than 10 genes have been identified, involving fibroblast growth factor, transforming growth factor beta, and Eph/ephrin signalling pathways. Mutations affect each human calvarial suture (coronal, sagittal, metopic, and lambdoid) differently, suggesting different gene expression patterns exist in each human suture. To better understand the molecular control of human suture morphogenesis we used microarray analysis to identify genes differentially expressed during suture fusion in children with craniosynostosis. Expression differences were also analysed between each unfused suture type, between sutures from syndromic and non-syndromic craniosynostosis patients, and between unfused sutures from individuals with and without craniosynostosis. RESULTS: We identified genes with increased expression in unfused sutures compared to fusing/fused sutures that may be pivotal to the maintenance of suture patency or in controlling early osteoblast differentiation (i.e. RBP4, GPC3, C1QTNF3, IL11RA, PTN, POSTN). In addition, we have identified genes with increased expression in fusing/fused suture tissue that we suggest could have a role in premature suture fusion (i.e. WIF1, ANXA3, CYFIP2). Proteins of two of these genes, glypican 3 and retinol binding protein 4, were investigated by immunohistochemistry and localised to the suture mesenchyme and osteogenic fronts of developing human calvaria, respectively, suggesting novel roles for these proteins in the maintenance of suture patency or in controlling early osteoblast differentiation. We show that there is limited difference in whole genome expression between sutures isolated from patients with syndromic and non-syndromic craniosynostosis and confirmed this by quantitative RT-PCR. Furthermore, distinct expression profiles for each unfused suture type were noted, with the metopic suture being most disparate. Finally, although calvarial bones are generally thought to grow without a cartilage precursor, we show histologically and by identification of cartilage-specific gene expression that cartilage may be involved in the morphogenesis of lambdoid and posterior sagittal sutures. CONCLUSION: This study has provided further insight into the complex signalling network which controls human calvarial suture morphogenesis and craniosynostosis. Identified genes are candidates for targeted therapeutic development and to screen for craniosynostosis-causing mutations.

Dataset Information

Visuomotor Function in School-Age Children with Single-Suture Craniosynostosis.

Publications

Visuomotor Function in School-Age Children with Single-Suture Craniosynostosis.

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