Unusual Giant Sialocyst Arising From Wharton's Duct: A Case Report
Maksym Skrypnyk, Margarita Skikevych, Roman Skrypnyk, Volodymyr Mykytenko, Igor Skrypnyk, Liudmyla Voloshyna, Tetiana Yatsenko, Chun Xu, Satoshi Takahashi, Taro Osada, Koichi Hattori, Beate HeissigABSTRACT
Background
Head and neck masses in adults are a common reason for referral to maxillofacial surgeons and otolaryngologists, and most are benign. True salivary duct cysts (sialocysts) are extremely rare, accounting for < 1% of salivary duct lesions, and primarily involve minor salivary glands. Involvement of major salivary glands is exceptional, with only isolated cases reported in the parotid gland. To the best of our knowledge, a sialocyst arising from the submandibular gland or Wharton's duct has not been reported.
Case Presentation
We report a unique case of a giant sialocyst originating from Wharton's duct and presenting as a large unilateral submandibular mass in a 58‐year‐old male. The patient presented with a slowly progressive, painless right submandibular swelling that enlarged rapidly over 4 months. Clinical examination revealed a well‐defined, asymptomatic mass with preserved salivary function. Computed tomography demonstrated a large, well‐encapsulated cystic lesion without inflammatory changes. Complete excision was achieved via an extraoral approach, with identification and ligation of a pathological communication with Wharton's duct. Aspiration revealed thick, viscous, mucin‐rich fluid. Histopathology confirmed a mucus retention cyst (sialocyst) with an epithelial lining.
Conclusion
This case expands the recognized anatomical distribution and clinical behaviour of sialocysts. It highlights the importance of including sialocysts in the differential diagnosis of cystic head and neck masses. It also underscores the diagnostic value of imaging and aspirate characteristics and demonstrates that complete cyst excision may be curative without requiring gland extirpation. Our findings emphasize that prior surgery or unintentional damage to ductal epithelial integrity are significant predisposing factors for the development of sialocysts.