Multidisciplinary Management of Hepatic Metastasis from Submandibular Gland Adenoid Cystic Carcinoma: A Case Report and Comprehensive Literature Review.

PURPOSE: Adenoid cystic carcinoma (ACC) of the submandibular gland is a rare malignancy characterized by an indolent clinical course and a high propensity for distant metastases. Hepatic metastases are particularly uncommon, and evidence guiding their optimal management is limited. This report aims to describe a rare case of isolated liver metastasis from submandibular gland ACC treated surgically and to contextualize it within the existing literature.

METHODS

We present the case of a 58-year-old woman who developed a solitary hepatic metastasis four years after primary surgical resection and adjuvant chemoradiotherapy for submandibular gland ACC. A multidisciplinary treatment strategy was adopted, including liver venous deprivation (LVD) to optimize the future liver remnant, followed by right hepatectomy. In addition, a comprehensive review of the literature was performed to identify all reported cases of surgically treated hepatic metastases from submandibular gland ACC.

RESULTS

The patient underwent successful right hepatectomy without major postoperative complications. Histopathological examination confirmed metastatic ACC with immunohistochemical features consistent with the primary tumor. At one year of postoperative follow-up, the patient remained disease-free. The literature review revealed marked heterogeneity in timing of metastasis, surgical approaches, and oncological outcomes, with variable disease-free survival and recurrence patterns following hepatic resection.

CONCLUSION

Surgical resection of isolated hepatic metastasis from submandibular gland ACC appears feasible and may provide durable disease control in carefully selected patients. A multidisciplinary approach, including preoperative liver hypertrophy techniques, can safely expand surgical indications. Given the potential for late metastatic recurrence, individualized treatment strategies and long-term follow-up are essential.