Sialodochostomy as treatment for imperforate submandibular duct: a systematic literature review and report of two cases.

Objective: To better understand the diagnosis, treatments, and outcomes of congenitally imperforate salivary ducts.

Methods: A systematic literature review was performed using the following terms: imperforate submandibular duct, imperforate Wharton's duct, congenital atresia submandibular duct, congenital atresia sublingual duct, and congenital ranula. Only papers focusing on congenital floor of mouth cysts arising from imperforate salivary ducts were included. Two additional patients from our own experience are also discussed.

Results: Seventeen papers published since 1950 met inclusion criteria. A total of 26 patients underwent therapy ranging from mere observation to complete excision of the cyst with the sublingual gland. Both of our 2 additional patients both underwent simple sialodochostomy (excision of the imperforate sublingual caruncle) with cyst decompression. For all 28 patients, there have been no reported recurrences, with mean follow-up of 24.8 months and median follow-up of 14.8 months.

Conclusions: Congenital floor of mouth cysts arising from imperforate salivary ducts are rare, but they may mimic the appearance of a ranula. Unlike ranulas, these cysts may be safely and effectively cured by simple sialodochostomy. To date, 26 other cases have been described in the literature, and nearly all have been successfully treated with simple incision or marsupialization without recurrence. Care should be taken to exclude ranula as a diagnosis, as incomplete excision may likely result in recurrence.