Kugimoto T, Yamagata Y, Ohsako T, Hirai H, Nishii N, Kayamori K, Ikeda T, Harada H. Massive low-grade myxoid liposarcoma of the floor of the mouth: A case report and review of literature. World J Clin Cases 2022; 10(34): 12742-12749 [PMID: 36579093 DOI: 10.12998/wjcc.v10.i34.12742]
Corresponding Author of This Article
Takuma Kugimoto, DDS, PhD, Assistant Professor, Department of Oral and Maxillofacial Surgical Oncology, Division of Oral Health Science, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, 1-5-45 Yushima, Bunkyo-ku 113-8549, Tokyo, Japan. kugimoto.osur@tmd.ac.jp
Research Domain of This Article
Oncology
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (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/
Takuma Kugimoto, Yuko Yamagata, Toshimitsu Ohsako, Hideaki Hirai, Naoto Nishii, Hiroyuki Harada, Department of Oral and Maxillofacial Surgical Oncology, Division of Oral Health Science, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Bunkyo-ku 113-8549, Tokyo, Japan
Kou Kayamori, Tohru Ikeda, Department of Oral Pathology, Division of Oral Health Science, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Bunkyo-ku 113-8549, Tokyo, Japan
Author contributions: Kugimoto T contributed to patient care, literature review, and drafting of the manuscript; Yamagata Y, Ohsako T, Hirai H, Nishii N, and Harada H contributed to patient care and editing of the manuscript; Kayamori K and Ikeda T contributed to pathologic analysis and editing of the manuscript; The final manuscript was reviewed and approved by all authors for submission; All authors read and approved the final manuscript.
Informed consent statement: Written informed consent was obtained from the patient for the publication of this case report and any accompanying images.
Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article.
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: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Takuma Kugimoto, DDS, PhD, Assistant Professor, Department of Oral and Maxillofacial Surgical Oncology, Division of Oral Health Science, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, 1-5-45 Yushima, Bunkyo-ku 113-8549, Tokyo, Japan. kugimoto.osur@tmd.ac.jp
Received: September 6, 2022 Peer-review started: September 6, 2022 First decision: October 12, 2022 Revised: October 20, 2022 Accepted: October 27, 2022 Article in press: October 27, 2022 Published online: December 6, 2022 Processing time: 86 Days and 19.8 Hours
Abstract
BACKGROUND
Oral liposarcoma is an extremely rare lesion that is often clinically misdiagnosed as a benign tumor due to its asymptomatic and indolent clinical course. Here, we report a case of massive low-grade myxoid liposarcoma (MLS) of the floor of the mouth.
CASE SUMMARY
A 71-year-old man presented with a huge mass in the left floor of the mouth. A biopsy was performed, and a diagnosis of a myxoid tumor suspicious for low-grade MLS or myxoma was made. Gadolinium-enhanced T1-weighted magnetic resonance imaging showed an intensely enhanced tumor lesion that occupies the left sublingual space and extends to the submandibular space. Submandibular dissection, tumor resection, and reconstruction with a radial forearm flap were performed. The surgical specimen exhibited histologically low-grade MLS. Fused in sarcoma (FUS, also known as TLS) and DNA damage-inducible transcript 3 (DDIT3, also known as CHOP) break-apart was not detected in the fluorescence in situ hybridization analysis. The tumor was completely encapsulated and did not require additional treatment. Furthermore, no recurrence was reported 40 mo after surgery.
CONCLUSION
We experienced an extremely rare, massive, low-grade MLS emerging from the floor of the mouth. Oftentimes, an MLS of the floor of the mouth lacks significant clinical findings and is often misdiagnosed. Although no FUS-DDIT3 fusion gene was detected, a low-grade MLS was ultimately diagnosed based on the histological findings.
Core Tip: Liposarcoma emerging from the floor of the mouth is extremely rare and often lacks significant clinical findings. This type of liposarcoma is usually misdiagnosed as ranula or benign tumor. In the present case, a definitive diagnosis could not be made preoperatively; however, myxoid liposarcoma was suggested, and complete resection could be achieved with appropriate imaging. A preoperative biopsy can help prevent incomplete resection. Clinical and radiological surveillance is necessary due to the possibility of local recurrence.
