Department of Dentistry and Oral Surgery, Unit of Sensory and Locomotor Medicine, Division of Medicine, Faculty of Medical Sciences, University of Fukui, Fukui, Japan.
Department of Thoracic Surgery, Unit of Surgery, Division of Medicine, Faculty of Medical Sciences, University of Fukui, Fukui, Japan.
Medicine (Baltimore). 2021 Dec 10;100(49):e28098. doi: 10.1097/MD.0000000000028098.
Adenoid 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.
A 52-year-old man was referred to our department with a 1-year history of painful swelling on the right oral floor.
An incisional biopsy was performed, and histopathological examination revealed malignancy.
Surgical 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.
After receiving palliative care, the patient died of multiple organ failure.
As 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.
腺样囊性癌（ACC）是一种罕见的恶性肿瘤，主要发生在唾液腺。尽管局部控制良好，但仍可能发生远处转移。此外，ACC 的远处转移可在无局部复发的长时间间隔后发生。我们报告首例舌下腺 ACC，其在初次治疗 20 年后发生肺转移。
一名 52 岁男性，因右口腔底部疼痛性肿胀 1 年就诊于我科。
进行了切开活检，组织病理学检查显示为恶性肿瘤。
行右口腔底部和右 supra-omohyoid 颈清扫术，并术后行放化疗，诊断为舌下腺 ACC。初次治疗 20 年后发现左肺转移。行转移瘤切除术，但随后出现皮肤和骨转移。
接受姑息治疗后，患者死于多器官衰竭。
由于唾液腺 ACC 的远处转移可能较晚发生，即使局部区域控制良好，接受初次治疗的患者也需要长期、严格的随访计划。
