In this study, we present a patient with a congenital cervical submandibular gland fistula who was admitted to the hospital for nodular thyroid hyperplasia. After bilateral subtotal thyroidectomy, the fistula tract and the submandibular salivary gland on the right side of the neck and the obliterated sinus on the left side of the neck were excised. The pathological examination led to the diagnosis of adenomatous hyperplasia of the thyroid gland with focal lymphocytic thyroiditis and mixed-type main salivary gland (submandibular gland) with cystic dilatation in one duct. Congenital salivary gland fistulae are rare and may be associated with abnormalities of the branchial apparatus or arise from aberrant or accessory salivary tissue. Excision of the fistula and the remaining submandibular gland is considered the treatment of choice in congenital fistulae, but traumatic fistulae generally resolve with conservative treatment within 3 weeks.

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