First branchial cleft cysts (FBCCs) are rare developmental anomalies that typically present in childhood. We report a unique case of a Type I FBCC in a 43-year-old male whose only symptom was isolated facial pain without infection or mass. Imaging and biopsies were inconclusive, prompting surgical excision for definitive diagnosis and symptomatic relief. Histopathology confirmed a branchial cleft anomaly. This case highlights the importance of including congenital anomalies in the differential diagnosis for atypical facial pain even in the adult population and underscores the value of a thorough and multi-disciplinary diagnostic workup.

First branchial cleft cyst

Congenital anomaly

Facial pain

Adult presentation

Misdiagnosis

© 2025 The Authors. Published by Elsevier Inc.