• Keiichi Ohta, Shinpei Matsuda, Akitoshi Okada, Masato Sasaki, Yoshiaki Imamura, and Hitoshi Yoshimura.
• Department of Dentistry and Oral Surgery, Unit of Sensory and Locomotor Medicine, Division of Medicine, Faculty of Medical Sciences, University of Fukui, Fukui, Japan.
• Medicine (Baltimore). 2021 Dec 10; 100 (49): e28098e28098.

RationaleAdenoid cystic carcinoma (ACC) is a rare malignant tumor that primarily occurs in the salivary glands. Distant metastases can develop despite favorable local control. Moreover, distant metastasis of ACC can occur after a long time interval without local recurrence. We report the first case of ACC of the sublingual gland that developed lung metastasis 20 years after primary treatment.Patient ConcernsA 52-year-old man was referred to our department with a 1-year history of painful swelling on the right oral floor.DiagnosisAn incisional biopsy was performed, and histopathological examination revealed malignancy.InterventionsSurgical excision of the right oral floor and right supra-omohyoid neck dissection with postoperative chemoradiation therapy were performed, and ACC of the sublingual gland was diagnosed. Left pulmonary metastasis was detected 20 years after the primary treatment. Metastasectomy was performed; however, subsequently, skin and bone metastases developed.OutcomesAfter receiving palliative care, the patient died of multiple organ failure.LessonsAs late distant metastasis of salivary ACC can develop, patients who undergo primary treatment need a long-term, strict follow-up plan even if locoregional control is favorable.Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.

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