Co-Presentation of Craniosynostosis and Ossified Cephalhematomas

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BACKGROUND: Sagittal craniosynostosis associated with ossified cephalhematomas has been reported, but whether this relationship is causal or coincidental remains unclear. It has been proposed that ossified cephalhematomas may increase the risk of premature suture fusion, or that craniosynostosis could predispose individuals to cephalhematoma formation or ossification. The authors evaluated the prevalence of cephalhematomas and craniosynostosis at their institution and reviewed cases where both coexisted. METHODS: The authors retrospectively reviewed craniosynostosis cases treated at a large children's hospital from November 2007 through September 2021. Epic SlicerDicer was used to identify instances of coexisting ossified cephalhematomas. Clinical features recorded included prenatal and postnatal course, size and location of ossified cephalhematoma, suture fused, interventions, and outcomes. RESULTS: Eight of 75 patients with ossified cephalhematomas had coexisting craniosynostosis. One was excluded because the cephalhematoma was contralateral to the synostosis, leaving seven patients (9.3%) with both conditions. Five had sagittal and two had right unicoronal synostosis. Deliveries included three spontaneous vaginal, three assisted vaginal (forceps or vacuum), and one cesarean. Cephalhematomas were recognized between birth and 96 days. All sagittal synostosis cases underwent successful sagittal craniectomy with biparietal morcellation, with removal of the cephalhematoma. Neither unicoronal case required surgery. CONCLUSIONS: In our series, 9.3% of ossified cephalhematoma cases had coexisting craniosynostosis. In most cases, the synostosis was sagittal, in accordance with the literature. The authors report the first two unicoronal craniosynostosis cases in the literature. There is a rare association between craniosynostosis and ossified cephalhematoma, but the nature of the relationship remains elusive.

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