Ghost cell odontogenic carcinoma arising in the background of a calcifying odontogenic cyst
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CASE REPORT
Ghost cell odontogenic carcinoma arising in the background of a calcifying odontogenic cyst Chané Nel1 · Liam Robinson1 · Willie F. P. van Heerden1 Received: 22 September 2020 / Accepted: 4 November 2020 © Japanese Society for Oral and Maxillofacial Radiology and Springer Nature Singapore Pte Ltd. 2020
Abstract Ghost cell odontogenic carcinoma (GCOC) is a rare malignant neoplasm, representing 3% of all ghost cell lesions of the jaws. They can arise de novo or from a pre-existing calcifying odontogenic cyst (COC) or dentinogenic ghost cell tumour (DGCT). A systematic review of the literature reported only 12 cases of a GCOC arising from a pre-existing COC. This report highlights an additional case of a GCOC arising from a pre-existing COC after 3 years in an adolescent male. The patient initially presented with a painless swelling of the right mandibular corpus. Panoramic radiographic examination showed an expansive unilocular radiolucent lesion. After 3 years, the radiographic features appeared more aggressive with increased expansion and cortical perforation. A wide surgical resection was performed, whereby the lesion was diagnosed as a GCOC. Due to the rarity of these malignant neoplasms, limited information is available regarding their biological behaviour. Oneyear follow-up revealed no clinical signs of recurrence. Keywords Odontogenic cysts · Odontogenic tumours · Ghost cell lesions of the jaws · Malignant transformation · Maxillofacial radiology
Introduction Ghost cell lesions of the jaws are rare, representing 0.4% of all head and neck lesions [1]. They may occur centrally within bone or in peripheral soft tissues. Much academic deliberation has occurred over the terminology of these ghost cell lesions due to their diverse biological behaviours. These lesions can occur as cystic, solid, or malignant tumours termed calcifying odontogenic cyst (COC), dentinogenic ghost cell tumour (DGCT), and ghost cell odontogenic carcinoma (GCOC), respectively. The 2017 World Health Organization (WHO) classification of head and neck tumours defines COC as a simple cyst lined by ameloblastoma-like epithelium containing localised accumulations of ghost cells [2]. The solid counterpart, termed DGCT, consists of infiltrating ameloblastomatous epithelium with varying degrees of basaloid cells. In addition, these tumours contain ghost cells and dentinoid material [2]. Malignant * Chané Nel [email protected] 1
Department of Oral Pathology and Oral Biology, Faculty of Health Sciences, University of Pretoria, Pretoria, South Africa
transformation of the aforementioned lesions may occur, resulting in an entity termed GCOC. This odontogenic carcinoma can also arise de novo [3], and is characterised by cytological evidence of malignancy and an infiltrative growth pattern with aberrant ghost cell keratinisation and dentinoid deposition [2]. GCOC represents 3% of all ghost cell lesions of the jaws with only limited cases reported in the literature [3, 4]. Ikemura et al. first described the neoplasm in 1985 as a
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