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February 7, 2011

Neurodevelopmental Outcomes in Children With Hemifacial Microsomia

Author Affiliations

Author Affiliations: Departments of Psychiatry and Behavioral Sciences (Drs Collett and Speltz) and Biostatistics (Dr Leroux), University of Washington, and Department of Psychiatry and Behavioral Medicine, Seattle Children's Hospital (Drs Collett and Speltz), Seattle, Washington; Department of Oral Biology, University of Pittsburgh School of Dental Medicine, Pittsburgh, Pennsylvania (Dr Cloonan); and Slone Epidemiology Center at Boston University, Boston, Massachusetts (Ms Kelly and Dr Werler).

Arch Pediatr Adolesc Med. 2011;165(2):134-140. doi:10.1001/archpediatrics.2010.271

Objective  To determine whether preadolescent children with hemifacial microsomia (HFM) have higher risk of neurodevelopmental delays than unaffected control individuals.

Design  Case-control follow-up study of neurodevelopment in children with and without HFM.

Setting  Case individuals were originally recruited from 26 craniofacial centers across the United States and Canada, and controls were recruited through community pediatricians.

Participants  One hundred thirty-six children with HFM (cases) and 568 unaffected children (controls).

Main Exposure  History of HFM.

Main Outcome Measures  The Peabody Picture Vocabulary Test–Third Edition, the Beery-Buktenica Developmental Test of Visual Motor Integration–Fifth Edition, and the Academic Competence scales from the Child Behavior Checklist and the Teacher Report Form.

Results  Children with HFM scored lower than controls on all measures (effect size = −0.27 to −0.45; P < .001 to P = .008). Compared with controls, cases were 2 to 3 times as likely to score in the at-risk range. Relative to controls, outcomes were worse for male cases and those whose mothers were 25 years or younger at the time of their birth. Cases with HFM plus other malformations had poorer outcomes, as did cases with hearing, vision, or speech impairments.

Conclusions  This is the first study, to our knowledge, to show that children with HFM have poorer neurodevelopmental outcomes than unaffected children, but further study using more detailed assessments is indicated. Clinically, the findings suggest that early neurodevelopmental screening is warranted for all children with HFM.