Little evidence of association between severity of trigonocephaly and cognitive development in infants with single-suture metopic synostosis.
OBJECTIVE
To measure severity of trigonocephaly among infants with single-suture metopic craniosynostosis by using a novel shape descriptor, the trigonocephaly severity index (TSI), and to evaluate whether degree of trigonocephaly correlates with their neurodevelopmental test scores.


METHODS
We conducted a multicenter cross-sectional and longitudinal study, identifying and recruiting 65 infants with metopic synostosis before their corrective surgery. We obtained computed tomography images for 49 infants and measured the presurgical TSI, a 3-dimensional outline-based cranial shape descriptor. We evaluated neurodevelopment by administering the Bayley Scales of Infant Development, Second Edition, and the Preschool Language Scale, Third Edition, before surgery and at 18 and 36 months of age. We fit linear regression models to estimate associations between test scores and TSI values adjusted for age at testing and race/ethnicity. We fit logistic regression models to estimate whether the odds of developmental delay were increased among children with more severe trigonocephaly.


RESULTS
We observed little adjusted association between neurodevelopmental test scores and TSI values, and no associations that persisted at 3 years. Trigonocephaly was less severe among children referred at older ages.


CONCLUSIONS
We observed little evidence of an association between the severity of trigonocephaly among metopic synostosis patients and their neurodevelopmental test scores. Detecting such a relationship with precision may require larger sample sizes or alternative phenotypic quantifiers. Until studies are conducted to explore these possibilities, it appears that although associated with the presence of metopic synostosis, the risk of developmental delays in young children is unrelated to further variation in trigonocephalic shape.
Starr
Jacqueline R
JR
Departments of Pediatrics and Epidemiology, University of Washington, Children's Craniofacial Center, Seattle Children's Hospital, Seattle, Washington, USA.
Lin
H Jill
HJ
Ruiz-Correa
Salvador
S
Cunningham
Michael L
ML
Ellenbogen
Richard G
RG
Collett
Brent R
BR
Kapp-Simon
Kathleen A
KA
Speltz
Matthew L
ML
D002652:Child Behavior; D002657:Child Development; D002675:Child, Preschool; D003072:Cognition Disorders / Q000209:etiology*; D003398:Craniosynostoses / Q000473:pathology* / Q000523:psychology* / Q000601:surgery; D003430:Cross-Sectional Studies; D005260:Female; D005500:Follow-Up Studies; D006801:Humans; D007223:Infant; D007804:Language Development; D016014:Linear Models; D008137:Longitudinal Studies; D008279:Magnetic Resonance Imaging; D008297:Male; D009483:Neuropsychological Tests; D019635:Neurosurgical Procedures; D011446:Prospective Studies; D014057:Tomography, X-Ray Computed