Adenoid Cystic Carcinoma With Sialolithiasis of the Left Submandibular Gland: A Case Report and Literature Review

Abstract

Adenoid cystic carcinoma is one of the most common salivary gland malignancies with poor long-term prognosis, but the coexistence of sialoliths is extraordinarily rare. In this article, we report a case of 30-year-old woman with a history of submandibular area swelling with intermittent pain increasing during mealtimes that had led her attending physician to diagnose a sialolith in the left submandibular gland on a radiograph 10 years before. However, the surgical specimen proved to be an adenoid cystic carcinoma accompanied with a sialolith. Histopathologically, the submandibular gland was displaced with a fibrous granulation tissue containing a small cribriform carcinoma invading the extracapsular region of the gland. We performed fluorescence in situ hybridization examination with an MYB-NFIB fusion probe of the lesion, with positive results. The patient underwent a supraomohyoid neck dissection as additional procedure because of the possibility of the extracapsular cancer nest remaining around the submandibular gland, but she remains well and disease free 11 years after the first operation.

Coexistence of Adenoid Cystic Carcinoma and Sialolithiasis in Submandibular Gland: Case Report

“…The duration of symptoms in some cases with the coexistence of sialolithiasis and salivary gland neoplasm in the literature is remarkable. The swellings duration was 10 years in Miyabe et al’s ( 9 ) and Hasegawa et al’s ( 4 ) cases, 3 years in Gallego et al’s ( 7 ) case, and 1.5 years in Nanda and Mehta's ( 10 ) case. In our study, as in these cases, the swelling was present for a long time (5 years).…”

Adenoid Cystic Carcinoma of Salivary Glands: A Ten-Year Review and an Assessment of the Current Management, Surgery, Radiotherapy, and Chemotherapy

“…Furthermore, SACC of the parotid has been reportedly associated with an odontogenic-like pain referring to the maxillary sinus and sialolithiasis [ 11 , 12 ]. Salivary adenoid cystic carcinoma of the submandibular gland can present as a slow-growing swelling in the floor of the mouth, often interfering with speech and mastication thus readily detectable or in the form of a lump affecting the submandibular area or presenting in the posterior lower border of the mandible on the affected side [ 13 ]. Other clinical features which have been linked to SACC of the submandibular gland include hyposalivation due to sublingual gland obstruction [ 14 ] and first bite syndrome, a sequela of parapharyngeal space surgery historically linked to the denervation of the parotid gland, not the submandibular [ 15 ].…”