A rare case of chronic genian fistula with oral mucosal hair follicle inclusion: case report and review of the literature

TL;DR

A rare case of a chronic cheek fistula with an unusual hair follicle inclusion is reported and discussed.

Contribution

This case report presents a rare clinical entity without prior facial surgery history.

Findings

A 73-year-old male had a chronic cutaneous fistula with intraoral hair follicle inclusion.

Surgical excision led to uneventful healing with no recurrence observed.

The case emphasizes the need for integrated clinical and diagnostic approaches for rare tracts.

Abstract

This report describes a rare case of chronic genian fistula associated with inclusion of oral mucosal hair follicle. Ectopic hair growth in the oral cavity is extremely rare and is typically associated with grafting techniques. The presence of a chronic extraoral fistula with intraoral hair follicle inclusion is exceedingly uncommon and poses diagnostic and therapeutic challenges.

A 73-year-old male presented to the Oral and Maxillofacial Surgery Department in Timișoara (Romania) with a chronic cutaneous fistula in the right lower cheek region, unresponsive to antibiotics and without signs of dental or sinus origin. Clinical examination revealed a 0.5 cm cutaneous opening and a filamentous, hair-like structure emerging intraorally from the right maxillary alveolar crest. A submucosal fibrotic tract was palpable, and although the fistula was not clearly visible on CBCT due to its soft tissue nature, imaging confirmed the absence of bony or odontogenic involvement. Given the clear clinical presentation, surgical excision was performed under local anesthesia. Intraoperatively, a keratinized, hair-bearing tract traversing the cheek to the alveolar process was excised en bloc. Healing was uneventful, and no recurrence was observed at 14 days.

This is one of the few reported cases of a genian fistula with intraoral hair follicle inclusion in a patient without prior facial surgery. The case highlights the importance of integrating clinical, radiologic, and histopathologic findings in diagnosing and managing rare fistulous tracts.

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