A case report: Congenital salivary fistula from the sublingual gland in an adult

Abstract

An unusual case of a congenital salivary fistula in an adult is reported. A 41-year-old woman presented with a clear, colorless, viscous, saliva-like discharge from a dimple on the submental skin. Fistulography with contrast dye demonstrated flow through the duct and showed that the contrast agent from the fistula went almost straight up to the floor of the oral cavity. A cavity about 7 mm in diameter was observed just before the end of the duct. This occurred without any flow into the oral cavity.With a clinical diagnosis of a congenital salivary fistula, complete excision of the fistula with the involved sublingual gland was performed under general anesthesia. The fistula penetrated the mylohyoid muscle, and part of it was seen to branch off into the sublingual gland in the middle of the fistula. On histopathological examination, the epithelium of the fistula was stratified squamous epithelium surrounded by skin tissue with hair follicles and sebaceous glands. At the end of the fistula, mucinous salivary gland tissue and lymphatic tissue with germinal centers were observed, and the salivary glands and fistula were continuous.Few adult cases of congenital salivary gland fistulas have been reported. This is the second case report of a congenital salivary gland fistula thought to originate from the sublingual gland. The lesion was completely excised, and the patient remains complication-free.