Coronal craniosynostosis of both the sporadic and syndromic types have been comprehensively described and extensively investigated. Previously, there have been no cases reported of acquired unilateral coronal craniosynostosis. We present a case of a 22-month-old male who developed a left unilateral coronal craniosynostosis following multiple surgical interventions for birth-related intracranial injuries. The genesis and molecular biology of craniosynostosis are discussed; patient presentation and treatment are reviewed.