Abstract
Wormian bones are accessory ossicles that typically occur within cranial sutures. Their presence within fontanelles, particularly concurrent with craniosynostosis, represents an uncommon finding that poses unique diagnostic and management considerations. We present a one-month-old male infant born at 35 weeks of gestation, who presented with an abnormal skull configuration. Physical examination revealed scaphocephaly with a palpable sagittal ridge, bilateral temporal hollowing, and concurrent distal shaft epispadias. Computed tomography (CT) with three-dimensional (3D) reconstruction demonstrated sagittal craniosynostosis with an unusually large wormian bone within the anterior fontanelle. The patient underwent successful surgical management with extensive craniectomy, calvarial reconstruction using cranial distractor springs, and concurrent epispadias repair. The coexistence of craniosynostosis with wormian bone in the anterior fontanelle is rarely reported in the literature. This case highlights the importance of comprehensive imaging evaluation in infants with abnormal skull morphology and emphasizes the role of CT with 3D reconstruction in surgical planning. Anterior fontanelle wormian bones may occur in association with craniosynostosis, requiring careful radiological assessment and multidisciplinary surgical management. Three-dimensional CT reconstruction provides essential anatomical detail for operative planning in complex calvarial anomalies.