Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Apr 26, 2021; 9(12): 2930-2936
Published online Apr 26, 2021. doi: 10.12998/wjcc.v9.i12.2930
Sclerosing polycystic adenosis of the submandibular gland: Two case reports
Leng Wu, Ying Wang, Chuan-Yu Hu, Chun-Ming Huang
Leng Wu, Chuan-Yu Hu, Chun-Ming Huang, Department of Stomatology, Tongji Hospital of Tongji Medical College of Huazhong University of Science and Technology, Wuhan 430000, Hubei Province, China
Ying Wang, Department of Pathology, Tongji Hospital of Tongji Medical College of Huazhong University of Science and Technology, Wuhan 430000, Hubei Province, China
Author contributions: Wu L and Wang Y collected, analyzed, and interpreted data and wrote the manuscript; Huang CM and Hu CY collected, analyzed, and interpreted data and critically reviewed the manuscript; All authors approved the final version of the manuscript.
Supported by National Natural Science Foundation of China, No. 82002893.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
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Corresponding author: Chun-Ming Huang, MD, PhD, Attending Doctor, Department of Stomatology, Tongji Hospital of Tongji Medical College of Huazhong University of Science and Technology, No. 1095 Jiefang Avenue, Qiaokou District, Wuhan 430000, Hubei Province, China. huangchunming89@whu.edu.cn
Received: January 9, 2021
Peer-review started: January 10, 2021
First decision: January 24, 2021
Revised: February 3, 2021
Accepted: February 24, 2021
Article in press: February 24, 2021
Published online: April 26, 2021
Abstract
BACKGROUND

Sclerosing polycystic adenosis (SPA) is a rare disease of salivary glands, similar to fibrocystic disease of the breast. It occurs over a wide age range and exhibits a slight female preference. Most SPA cases have occurred in the parotid gland. The exact nature of SPA is unclear, but its tumor nature has recently been proposed. Although SPA has a good prognosis after adequate surgery, atypical lesions might occur, ranging from mild dysplasia to carcinoma in situ in some cases. To the best of our knowledge, only five cases of SPA in the submandibular gland have been reported to date. Here, we present two new cases of SPA involving the submandibular gland.

CASE SUMMARY

A 50-year-old woman and a 52-year-old woman were referred to Tongji Hospital in Wuhan, China, with complaints of moderate pain, recurrent swelling, and a mass in the submandibular area. After admission, the two cases of the submandibular mass were examined physically. The boundary of the submandibular tumor was clear, and the range of motion was good. After preoperative examinations, surgery was performed on a selective basis. Postoperative histopathological examination revealed a well-defined mass with acinar structures, ducts, or cystic dilated glands of various sizes scattered in a large number of proliferative sclerosing stroma. There were flat and cuboidal cells, and eosinophils in the duct epithelium. There was also a eosinophilic substance in the lumen of dilated cysts. No atypical epithelial hyperplasia, invasive growth, or carcinoma in situ was found. Based on the above findings, the mass was diagnosed as SPA. Both patients have remained asymptomatic and no recurrence or distant metastasis had occurred by the 7-mo and 5-year follow-up, respectively.

CONCLUSION

SPA is a rare disease of the salivary gland. Even though it has a good prognosis after adequate surgery, atypical lesions may occur from mild dysplasia to carcinoma in situ. However, no recurrence, distant metastasis, or mortality has been reported for submandibular gland SPA. Clinicians and pathologists should be familiar with the characteristics of SPA in the submandibular gland to avoid misdiagnosis and overtreatment.

Keywords: Sclerosing polycystic adenosis, Submandibular gland, Histopathology, Diagnosis, Treatment, Case report

Core Tip: Sclerosing polycystic adenosis (SPA) is a rare disease of the salivary gland. To the best of our knowledge, only 5 cases of SPA in the submandibular gland have been reported. Here, we present two new cases of SPA involving the submandibular gland. Even though SPA has a good prognosis after adequate surgery, atypical lesions may occur from mild dysplasia to carcinoma in situ. To date, there have been no reports of recurrence, distant metastases, or mortality associated with SPA in the submandibular gland. Clinicians and pathologists should be familiar with the characteristics of this disease to avoid misdiagnosis and overtreatment.