Transformation of an odontogenic keratocyst into a solid variant of odontogenic keratocyst/keratoameloblastoma during long‑term follow‑up: A case report

Yamasaki,Sachiko; Shintani,Tomoaki; Ando,Toshinori; Miyauchi,Mutsumi; Yanamoto,Souichi

doi:10.3892/mmr.2024.13168

Transformation of an odontogenic keratocyst into a solid variant of odontogenic keratocyst/keratoameloblastoma during long‑term follow‑up: A case report

Authors:
- Sachiko Yamasaki
- Tomoaki Shintani
- Toshinori Ando
- Mutsumi Miyauchi
- Souichi Yanamoto
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Affiliations: Department of Oral Oncology, Graduate School of Biomedical and Health Sciences, Hiroshima University, Hiroshima 734‑8553, Japan, Center of Oral Clinical Examination, Hiroshima University Hospital, Hiroshima 734‑8553, Japan, Department of Oral and Maxillofacial Pathobiology, Graduate School of Biomedical and Health Sciences, Hiroshima University, Hiroshima 734‑8551, Japan
Published online on: January 19, 2024 https://doi.org/10.3892/mmr.2024.13168
Article Number: 44
Copyright: © Yamasaki et al. This is an open access article distributed under the terms of Creative Commons Attribution License.

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Abstract

Keratoameloblastoma (KA) and solid variant of odontogenic keratocyst (SOKC) are rare odontogenic lesions, and their relationship and differences are unclear. The present study described a case that started as an odontogenic keratocyst (OKC) and transformed to SOKC/KA upon recurrence. Briefly, a 26‑year‑old man presented with swelling in the right cheek and was referred to the Department of Oral and Maxillofacial surgery, Hiroshima University Hospital (Hiroshima, Japan). At the initial visit, unicystic bone permeation was observed extending from the right canine to the molar, maxillary sinus and nasal cavity. After the biopsy, the patient underwent excisional surgery and was diagnosed with OKC. Thereafter, the lesion recurred six times over a period of 13 years and showed different histopathological features from those of the primary lesion, all consisting of numerous cysts with keratinization, which were diagnosed as SOKC/KA. The Ki‑67 positivity rate was ~10%, which was higher than that of the primary lesion, but there was no atypia. Genetic analysis of the recurrent lesion revealed mutations in adenomatous polyposis coli and Kirsten rat sarcoma viral oncogene homolog. This case originated from OKC, and the morphological features of OKC and KA were mixed upon recurrence, supporting the commonality and association between the two. However, multiple mutations different from those of OKC and ameloblastoma were detected, suggesting an association of SOKC/KA with increased proliferative activity and a high recurrence rate.

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