Orocutaneous fistula with oral mucosal hair growth: A rare presentation

Dear Editor,

Hair growth in the oral mucosa following reconstruction surgery using skin grafts containing intact hair follicles has been well documented in the literature. We report an unusual case of an orocutaneous fistula with intraoral hair growth, occurring without any prior surgical intervention or skin grafting.

A 67-year-old male patient reported with a history of pain and swelling on his gums of one week duration. His vital signs were within normal limits. On extra-oral examination, a deep pit was noted on the skin near the right commissure 2 cm above the lower border of the mandible suggestive of a fistula. His oral hygiene was poor with multiple mobile teeth as a result of chronic generalised periodontitis. A few strands of hair were observed in the buccal vestibule adjacent to the edentulous right lower premolar region [Figure 1]. The intra-oral opening of the suspected fistula was also evident in that region.

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Figure 1:

Intraoral hair growth in the right buccal vestibule (yellow arrow).

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On further questioning, the patient gave a history of a road traffic accident 25 years ago, resulting in the loss of several anterior teeth. Nearly 10 years ago, he underwent the extraction of a decayed right mandibular posterior tooth which was accompanied by swelling and pus discharge.

The patient believed that the hair-like strands were remnants of suture threads from a previous tooth extraction and, since they caused no symptoms, did not seek treatment until now. The persistent cutaneous fistula may have resulted from tissue damage sustained during a previous road traffic accident or as a complication following a draining dentoalveolar abscess. A panoramic radiograph with fistula tracing was obtained to confirm the presence of a persistent orocutaneous fistula. The patency of the fistula was evident in the radiograph [Figure 2]. The treatment plan consisted of the removal of hair strands along with fistulectomy and total extraction of teeth followed by prosthetic rehabilitation.

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Figure 2:

Fistula tracing using Gutta-Percha cone in panoramic radiograph (yellow arrow).

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Even though the presence of ectopic sebaceous glands in oral mucosa is common, the occurrence of hair alongside is extremely rare. Apart from patients with a history of surgical skin grafts, only four case reports of hair growth in the oral mucosa with unclear aetiopathogenesis have been found in the literature.^1-4 Researchers suggested heterotopia of the pilosebaceous unit as the most likely explanation of such occurrences. Intra-oral hair can result in emotional and psychological distress to the patient. The excessive hair in the oral cavity causes difficulty in eating, swallowing, and speech.⁵

As our patient had a history of a road traffic accident and alveolar abscess with persistent orocutaneous fistula, we suggest accidental displacement of skin appendage into the oral cavity following a road traffic accident, skin appendage formation during epithelisation of the fistulous tract, and pilosebaceous metaplasia of oral mucosa as some of the possibilities for the occurrence of hair in the oral mucosa. Moreover, such occurrence of hair growth in oral mucosa associated with persistent orocutaneous fistula has not yet been reported to the best of our knowledge.