A Rare Cheek Sinus Presentation of a Type II First Branchial Cleft Anomaly: A Case Report and Literature Review

Background: Congenital cheek sinuses are exceedingly rare craniofacial anomalies, typically present at birth and often misdiagnosed due to their resemblance to dermal pits or dimples. Only a few cases have been reported in the literature. This case is notable for its histopathological confirmation as a type II first branchial cleft anomaly containing an accessory tragus, highlighting the embryologic and clinical overlap between congenital cheek sinuses and branchial cleft anomalies. Case presentation: We report a three-year-old boy presenting with a congenital dimple along the right nasolabial fold. The lesion was present since birth and occasionally discharged clear fluid but had no history of infection or inflammation. Imaging demonstrated a subcutaneous tract without communication to the oral cavity or parotid gland. Under general anesthesia, surgical excision was performed, and the sinus tract was dissected in continuity to a cartilaginous remnant. Histopathologic analysis confirmed the presence of an accessory tragus consistent with a type II first branchial cleft anomaly. The patient’s postoperative recovery was uneventful, and at three months’ follow-up, there was no recurrence and an excellent cosmetic outcome. Conclusions: This case emphasizes the importance of considering branchial cleft anomalies in the differential diagnosis of congenital cheek sinuses. Although these lesions may appear benign and superficial, histopathological confirmation is critical for accurate classification. Complete surgical excision remains the definitive treatment, ensuring both diagnostic clarity and favorable aesthetic results. Increased awareness of such rare anomalies can improve diagnostic accuracy and surgical planning, particularly with respect to their potential proximity to critical facial structures.