Revisiting a Rare Cause of Plagiocephaly: Frontosphenoidal Craniosynostosis.

Frontosphenoidal craniosynostosis remains a rare cause of anterior plagiocephaly, with only 49 cases reported worldwide. Delayed diagnosis may lead to significant aesthetic and functional consequences. This case series presents 2 recently treated patients, highlighting clinical features, imaging findings, and surgical management. A retrospective review of 2 patients diagnosed and treated at our multidisciplinary head shape clinic was conducted, with parental consent obtained. Both children presented with unilateral brow flattening and preserved brow height. Imaging confirmed frontosphenoidal suture fusion with right frontal bone and supraorbital ridge flattening. Due to the severity of deformity, both underwent fronto-orbital advancement (FOAR) with the affected side bandeau and frontal bone removed, remodeled, and advanced to match the contralateral side. Both were discharged on postoperative day 4 with no complications and demonstrated marked cosmetic improvement. Multidisciplinary assessment and surgical intervention with FOAR led to improved aesthetic outcomes and avoided immediate functional deficits in our patients.