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Review
. 2021 Dec 10;100(49):e28098.
doi: 10.1097/MD.0000000000028098.

Adenoid cystic carcinoma of the sublingual gland developing lung metastasis 20 years after primary treatment: A case report and literature review

Keiichi Ohta  1 , Shinpei Matsuda  1 , Akitoshi Okada  2 , Masato Sasaki  2 , Yoshiaki Imamura  3 , Hitoshi Yoshimura  1
Affiliations
Review

Adenoid cystic carcinoma of the sublingual gland developing lung metastasis 20 years after primary treatment: A case report and literature review

Keiichi Ohta et al. Medicine (Baltimore). .

Abstract

Rationale: 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.

Patient concerns: A 52-year-old man was referred to our department with a 1-year history of painful swelling on the right oral floor.

Diagnosis: An incisional biopsy was performed, and histopathological examination revealed malignancy.

Interventions: 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.

Outcomes: After receiving palliative care, the patient died of multiple organ failure.

Lessons: 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.

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Conflict of interest statement

The authors have no conflicts of interest to disclose.

Figures

Figure 1
Figure 1
Intraoral examination showing a solitary, elastic hard, spherical 45 × 30 mm submucosal mass and paresthesia on the right oral floor.
Figure 2
Figure 2
The resected specimen showing a solid mass with surrounding soft tissues. The mass was filled with white, uniform material.
Figure 3
Figure 3
Histopathological findings showing islands of basaloid cells surrounding cyst-like spaces (pseudocysts), revealing a cribriform pattern, hematoxylin and eosin stain; magnification, × 100.
Figure 4
Figure 4
Chest radiograph showing a rounded mass shadow in the left lower lobe.
Figure 5
Figure 5
Axial chest computed tomography (CT) (lung window setting) showing 8.5 cm mass shadow in the left lower lobe, and many flat nodules along with the left pleura.
Figure 6
Figure 6
Histopathological findings of resected lung tumor showing a histological pattern similar to the sublingual gland, excised 20 years back, hematoxylin and eosin stain; magnification, × 100.
See this image and copyright information in PMC

References

    1. Coca-Pelaz A, Rodrigo JP, Bradley PJ, et al. . Adenoid cystic carcinoma of the head and neck – An update. Oral Oncol 2015;51:652–61. - PubMed
    1. Sentani K, Ogawa I, Ozasa K, et al. . Characteristics of 5015 salivary gland neoplasms registered in the Hiroshima tumor tissue registry over a period of 39 years. J Clin Med 2019;8:566. - PMC - PubMed
    1. Ko YH, Lee MA, Hong YS, et al. . Prognostic factors affecting the clinical outcome of adenoid cystic carcinoma of the head and neck. Jpn J Clin Oncol 2007;37:805–11. - PubMed
    1. Bjørndal K, Krogdahl A, Therkildsen MH, et al. . Salivary gland carcinoma in Denmark 1990-2005: a national study of incidence, site and histology. Results of the Danish Head and Neck Cancer Group (DAHANCA). Oral Oncol 2011;47:677–82. - PubMed
    1. Atallah S, Casiraghi O, Fakhry N, et al. . A prospective multicentre REFCOR study of 470 cases of head and neck adenoid cystic carcinoma: epidemiology and prognostic factors. Eur J Cancer 2020;130:241–9. - PubMed

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