Case Report
Copyright ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jul 6, 2019; 7(13): 1686-1695
Published online Jul 6, 2019. doi: 10.12998/wjcc.v7.i13.1686
Chronic progression of recurrent orthokeratinized odontogenic cyst into squamous cell carcinoma: A case report
Ruo-Yi Wu, Zhe Shao, Tian-Fu Wu
Ruo-Yi Wu, Zhe Shao, Tian-Fu Wu, The State Key Laboratory Breeding Base of Basic Science of Stomatology (Hubei-MOST) and Key Laboratory of Oral Biomedicine Ministry of Education, School and Hospital of Stomatology, Wuhan University, Wuhan 430072, Hubei Province, China
Ruo-Yi Wu, Zhe Shao, Tian-Fu Wu, Department of Oral and Maxillofacial Surgery, School and Hospital of Stomatology, Wuhan University, Wuhan 430072, Hubei Province, China
Author contributions: Wu RY and Shao Z contributed equally to this work; Wu RY and Shao Z collected the information and reviewed the literature; Wu TF and Wu RY wrote the paper.
Supported by Wuhan Morning Light Plan of Youth Science and Technology, No. 2017050304010305; National Natural Science Foundation of China, No. 81702705.
Informed consent statement: The patient involved was informed prior to submission of this case report and provided written consent.
Conflict-of-interest statement: We have no conflicts of interest to declare.
CARE Checklist (2016) statement: CARE Checklist (2016) statement has been uploaded.
Open-Access: 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/
Corresponding author: Tian-Fu Wu, PhD, Attending Doctor, Research Associate, Surgical Oncologist, Department of Oral and Maxillofacial Surgery, School and Hospital of Stomatology, Wuhan University, School and Hospital of Stomatology, Wuhan University, Luo Yu Road No. 237, Wuhan 430072, Hubei Province, China. wutianfu@whu.edu.cn
Telephone: +86-27-87873260 Fax: +86-27-87873260
Received: February 13, 2019
Peer-review started: February 15, 2019
First decision: March 9, 2019
Revised: March 27, 2019
Accepted: May 1, 2019
Article in press: May 1, 2019
Published online: July 6, 2019
Abstract
BACKGROUND

Orthokeratinized odontogenic cyst (OOC) is a benign odontogenic cyst. It is a variant of the common odontogenic keratocyst (OKC). This case report describes a rare malignant transformation of OOC, with the aim of raising awareness of the malignant potential of OOC and distinguishing it from OKC.

CASE SUMMARY

In August 2018, a 52-year-old man was referred to the Department of Oral Maxillofacial and Head–Neck Oncology of Wuhan University. The patient presented with severe pain in the left mandible for 2 mo, and had a 5-year history of osteomyelitis and mandibular cyst with three recurrences. His latest diagnosis by pathological examination was OOC of the left mandible with mild-to-moderate local proliferation. However, the cyst showed malignant potential by radiographic examination. We performed partial mandibulectomy and sent the lesion tissue for pathological examination. As expected, the cyst had deteriorated to moderately differentiated squamous cell carcinoma. During postoperative follow-up, the patient went for chemotherapy in September 2018 and successfully completed four cycles.

CONCLUSION

Surgeons should be more aware of OOC, which is usually benign but can become malignant.

Keywords: Orthokeratinized odontogenic cyst, Cancerization, Cancer recurrence, Squamous cell carcinoma, Odontogenic keratocyst, Case report

Core tip: Orthokeratinized odontogenic cyst (OOC) is a benign odontogenic cyst that rarely undergoes malignant transformation. In order to highlight the malignant potential of OOC, especially in patients with a history of recurrence and inflammation, we describe a rare case of recurrent OOC that transformed into squamous cell carcinoma after the patient underwent curettage twice.