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): 2908-2915
Published online Apr 26, 2021. doi: 10.12998/wjcc.v9.i12.2908
Salivary duct carcinoma of the submandibular gland presenting a diagnostic challenge: A case report
Toshihiro Uchihashi, Shingo Kodama, Akinari Sugauchi, Shinichiro Hiraoka, Katsutoshi Hirose, Yu Usami, Susumu Tanaka, Mikihiko Kogo
Toshihiro Uchihashi, Shingo Kodama, Akinari Sugauchi, Shinichiro Hiraoka, Mikihiko Kogo, First Department of Oral and Maxillofacial Surgery, Osaka University Graduate School of Dentistry, Suita 565-0871, Osaka, Japan
Katsutoshi Hirose, Yu Usami, Department of Oral Pathology, Osaka University Graduate School of Dentistry, Suita 565-0871, Osaka, Japan
Susumu Tanaka, First Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, Osaka University, Suita 565-0871, Osaka, Japan
Author contributions: Uchihashi T, Kodama S, Sugauchi A, Hiraoka S, and Tanaka S oversaw patient care; Uchihashi T and Kodama S wrote the manuscript; Hirose K and Usami Y performed the histopathological analysis; Kogo M and Tanaka S finalized the manuscript; all authors have read and approved the final manuscript.
Informed consent statement: Written informed consent was obtained from the patient’s legal guardian and from the patient for publication of this case report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no competing interests.
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).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Susumu Tanaka, DDS, PhD, Associate Professor, First Department of Oral and Maxillofacial Surgery, Graduate School of Dentistry, Osaka University, 1-8 Yamadaoka, Suita 565-0871, Osaka, Japan. stanaka@dent.osaka-u.ac.jp
Received: December 10, 2020
Peer-review started: December 10, 2020
First decision: January 29, 2021
Revised: February 9, 2021
Accepted: March 3, 2021
Article in press: March 3, 2021
Published online: April 26, 2021
Processing time: 125 Days and 17.6 Hours
Abstract
BACKGROUND

Salivary duct carcinoma (SDC) is a rare, extremely aggressive malignancy that arises in the submandibular gland. It can metastasize locally early and therefore is an important differential diagnosis of metastatic disease in cervical lymph nodes or specific lymphadenitis such as tuberculous cervical lymphadenitis.

CASE SUMMARY

We report a case of SDC in the submandibular gland that presented diagnostic difficulty. The lesion was coincidentally discovered through examination of the radiolucent area of the maxilla. Imaging failed to confirm the possibility of specific inflammation, leading us to execute an open biopsy to verify the diagnosis. The surgical specimen showed that the submandibular gland was primarily replaced with a calcified body. Following histological analysis and confirmation, we performed surgical resection, radiotherapy, and various chemotherapies.

CONCLUSION

Radiographic imaging characteristics of lymph node metastases of salivary gland cancer, especially of SDC, may resemble other cervical lymphadenitis; calcification at the submandibular gland is the landmark of SDC occurring at the subman-dibular gland.

Keywords: Salivary duct carcinoma; Tuberculous cervical lymphadenitis; Submandibular gland; Calcification; Salivary cancers; Chemotherapy; Case report

Core Tip: Salivary duct carcinoma (SDC) arising in the submandibular gland is particularly rare, and the related lymph node metastasis is difficult to distinguish from tuberculous cervical lymphadenitis, which complicates the establishment of a definitive diagnosis. We report our experience with a case of SDC in the submandibular gland, in which the similarities in the radiographic and imaging characteristics of lymph node metastatic SDC indeed mimicked those of tuberculous cervical lymphadenitis. However, calcification at the submandibular grand proved to be a critical finding that enabled a definitive diagnosis.