Congenital anterior cervical fistula originating from the Wharton duct

A 14-year-old boy presented with a spontaneous anterior cervical fistula, which exuded a saliva-like fluid during eating. Cervical examination revealed a hole 7 mm posterior to the hyoid bone, 5mm left of the median. Magnetic resonance imaging showed a low-intensity spindly mass with high-intensity margins. The mass was connected to the left submandibular gland. Contrast radiography of the fistula using Gastrografin demonstrated an ascending fistula that extended to the submandibular area. Intraoperatively, the fistula was shown to have ascended superficial to the level of the anterior cervical muscles, and it formed a cyst anterior to the hyoid bone. It then continued to the left submandibular gland. The fistula and submandibular gland were removed together. Wharton's duct was not found, and the orifice of the duct was unclear. Pathological examination of the fistula showed the lining epithelium at the side of the submandibular gland to be composed of columnar epithelial cells with stratified squamous cells at the epidermal side. Several acinar systems were found along the orifice of the fistula. There have been no previous reports of the Wharton duct running from the submandibular gland to the anterior cervical skin.