Case Report
Copyright ©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jun 6, 2020; 8(11): 2374-2379
Published online Jun 6, 2020. doi: 10.12998/wjcc.v8.i11.2374
Reduced delay in diagnosis of odontogenic keratocysts with malignant transformation: A case report
Xiao-Juan Luo, Ming-Liang Cheng, Chun-Ming Huang, Xiao-Ping Zhao
Xiao-Juan Luo, Ming-Liang Cheng, Chun-Ming Huang, Xiao-Ping Zhao, Center of Stomatology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430030, Hubei Province, China
Author contributions: Zhao XP contributed to study design, data collection and analysis, and manuscript preparation; Cheng ML, Luo XJ, and Huang CM contributed to data collection and analysis and manuscript preparation; Luo XJ and Cheng ML contributed equally to this article.
Supported by the National Natural Science Foundation of China, No. 81600911.
Informed consent statement: Informed written consent was obtained from the patient.
Conflict-of-interest statement: All authors declare no competing non-financial 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: Xiao-Ping Zhao, MD, PhD, Chief Doctor, Center of Stomatology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, No. 1095, Jiefang Avenue, Hankou, Wuhan 430030, Hubei Province, China. yinzikou@hotmail.com
Received: January 13, 2020
Peer-review started: January 13, 2020
First decision: April 27, 2020
Revised: April 27, 2020
Accepted: April 30, 2020
Article in press: April 30, 2020
Published online: June 6, 2020
Processing time: 146 Days and 13.3 Hours
Abstract
BACKGROUND

In rare cases, odontogenic keratocysts (ODs) transform into squamous cell carcinoma. Intervals between the first attendance of a patient and the diagnosis of OD with malignant transformation vary from weeks to years. In this article, we report a case of malignancy derived from OD with a five-day delay in diagnosis.

CASE SUMMARY

A 54-year-old woman was referred to Tongji Hospital in Wuhan, China with complaints of moderate pain, recurrent swelling, and pus discharge around her left maxillary lateral incisor for over 10 years. Physical examination revealed a fistula at the palatine-side mucoperiosteum of the left maxillary lateral incisor and enlarged lymph node in the left neck. Cone beam computed tomography revealed a cystic lesion with massive bone destruction from the left maxillary central incisor to the left secondary maxillary premolar and local bony destruction in the left first mandibular molar. The patient was clinically diagnosed with OD. Enucleation rather than marsupialization was performed given the risk factors of long history, recent aggravated pain, and massive bony destruction. Malignant transformation of OD was confirmed by pathologists 3 d after the operation. Radical surgery was performed, and lymph node metastasis was observed. The patient was subjected to postoperative radiotherapy and synchronous chemotherapy, and no local recurrence or distant metastasis was noted at one-year follow-up.

CONCLUSION

Our case suggests that clinicians should be aware of the malignant transformation of OD, especially when patients present with a long history, massive cyst, chronic inflammation, recent persistent infections, aggravated pain, numbness around the cystic lesion, and lymph node enlargement.

Keywords: Odontogenic keratocysts; Malignant transformation; Primary intraosseous squamous cell carcinoma; Delay in diagnosis; Enucleation; Case report

Core tip: We report a rare case of odontogenic keratocyst with malignant transformation. Although distinguishing between a benign and malignant odontogenic keratocyst is clinically challenging, factors, such as long history, massive cyst, chronic inflammation, recent persistent infections, aggravated pain, numbness around the cystic lesion, and lymph node enlargement, may suggest an overlooked malignant transformation.