Craniofacial Dysmorphology in Infants with Non-Syndromic Unilateral Coronal Craniosynostosis

Christopher P. Bellaire, Alex Devarajan, James G. Napoli, John W. Rutland, Helen Liu, Laya Jacob, Max Mandelbaum, Farah Sayegh, Ilana G. Margulies, Pedram Goel, Bradley N. Delman, Mark M. Urata, Peter J. Taub

Research output: Contribution to journalArticlepeer-review


Background: Unilateral coronal craniosynostosis (UCS) is a congenital disorder resulting from the premature suture fusion, leading to complex primary and compensatory morphologic changes in the shape of not only the calvarium and but also into the skull base. This deformity typically requires surgery to correct the shape of the skull and prevent neurologic sequelae, including increased intracranial pressure, sensory deficits, and cognitive impairment. Methods: The present multicenter study sought to reverse-engineer the bone dysmorphogenesis seen in non-syndromic UCS using a geometric morphometric approach. Computed tomography scans for 26 non-syndromic UCS patients were converted to three-dimensional mesh models. Two hundred thirty-six unique anatomical landmarks and semi-landmarked curves were then plotted on each model, creating wireframe representations of the Patients skulls. Results: Generalized Procrustes superimposition, Principal Component Analysis, and heatmaps identified significant superior displacement of the ipsilateral orbit ("harlequin" eye deformity), anterior displacement of the ear ipsilateral to the fused coronal suture, acute deviation of midline skull base structures ipsilateral to the fused coronal suture and flattening of the parietal bone and associated failure to expand superiorly. Conclusions: The described technique illustrates the impact of premature coronal suture fusion on the development of the entire skull and proposes how bone dysmorphology contributes to the Patients neurologic sequelae. By bridging novel basic science methodologies with clinical research, the present study quantitatively describes craniofacial development and bone dysmorphogenesis.

Original languageEnglish
Pages (from-to)1903-1908
Number of pages6
JournalJournal of Craniofacial Surgery
Issue number6
StatePublished - 1 Sep 2022


  • Cranial sutures
  • craniosynostosis
  • diseases
  • infant
  • neurosurgery
  • newborn
  • plastic
  • skull base
  • surgery


Dive into the research topics of 'Craniofacial Dysmorphology in Infants with Non-Syndromic Unilateral Coronal Craniosynostosis'. Together they form a unique fingerprint.

Cite this