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A congenital cheek fistula is a rare malformation in the buccal area. Here, we report the case of a congenital cheek fistula in a 50-year-old woman who visited our clinic with complaints of swelling and pain in her left cheek. Physical examination revealed a small hole in the left corner of the mouth present since birth. She had no other congenital malformations in the maxillofacial region such as an accessory ear and cleft lip. Manual compression of the cheek mass induced serous discharge from the hole. Magnetic resonance imaging (MRI) showed a cystic lesion in the left cheek and a fistula within the orbicularis oris muscle that opened into the small hole. After immediate incision and drainage of the cyst, both the cyst and fistula were surgically resected. The cystic lesion was completely delineated from the boundary of the parotid gland. The orbicularis oris muscle was partially incised to remove the fistula and the surrounding scar tissue. Histopathological examination of the resected specimen revealed a cavity consisting of epithelium inside the fistula. The postoperative course was insignificant. No recurrence of the cyst was observed six months postoperatively. The operative and pathological findings demonstrated that the ectoderm-derived epithelial tissue was enclosed by the mesoderm-derived muscle tissue. The mixture of different germ layer-derived tissues suggested that the fistula was a type of congenital transverse facial cleft induced by malfusion of the mandibular and maxillary prominences during embryonic development. The differential diagnoses of the congenital cheek fistula included orocutaneous fistulas and salivary fistulas. MRI was useful in delineating the border between the lesion and the surrounding tissue.

Congenital Cheek Fistula: A Rare Case of Developmental Abnormality in the Buccal Area