Glandular Odontogenic Cyst with Metaplastic Cartilage: Report of an Unusual Case and Literature Review
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CASE REPORTS
Glandular Odontogenic Cyst with Metaplastic Cartilage: Report of an Unusual Case and Literature Review Hannah Crane1 · Bhavesh Karbhari2 · David Hughes3 · Robert Orr2 · Daniel Brierley1 Received: 18 August 2020 / Accepted: 15 October 2020 © The Author(s) 2020
Abstract Glandular odontogenic cysts are rare odontogenic cysts with a wide range of histopathological features. In this paper we describe the clinical and pathological features of an unusual case of a glandular odontogenic cyst with metaplastic cartilage. The previous literature of odontogenic cysts presenting with metaplastic cartilage is reviewed alongside a discussion of the differential diagnoses. To our knowledge this is the first reported case of a glandular odontogenic cyst with metaplastic cartilage. Keywords Glandular odontogenic cyst · Odontogenic cysts · Diagnosis · Pathology
Introduction Glandular odontogenic cyst was first described by Padayachee and van Wyk in 1987 as a “sialo-odontogenic cyst”[1]. Gardner et al. [2] re-named the cyst as a glandular odontogenic cyst (GOC) due to the lack of evidence of a salivary origin and this nomenclature was subsequently accepted by the World Health Organisation [3]. GOC is a rare odontogenic cyst, with a demographic study showing they only account for 0.2% of odontogenic cysts within a UK population [4]. It occurs over a wide age range, with most cases diagnosed in the 5–7th decade with no gender predilection [5]. It commonly presents as a unilocular or multilocular radiolucency and is more frequently seen in the mandible [5, 6], with some studies showing a higher prevalence in the anterior regions of the jaws [5]. There are a wide range of histopathological features. Fowler et al. described 10 microscopic findings that could aid in diagnosis of GOC [5]. They suggested, following statistical analysis, that the * Hannah Crane [email protected] 1
Academic Unit of Oral and Maxillofacial Medicine and Pathology, School of Clinical Dentistry, 19 Claremont Crescent, Sheffield, UK
2
Department of Oral and Maxillofacial Surgery, Chesterfield Royal Hospital, Chesterfield, UK
3
Department of Histopathology, Sheffield Teaching Hospitals NHS Foundation Trust, Sheffield, UK
diagnosis can be confidently made when 7 out of the 10 following features are present; eosinophilic “hobnail” cells, apocrine metaplasia, intra-epithelial microcysts, variable thickness of the epithelial lining, clear cells in the basal layer, papillary projections, cilia, multiple cystic compartments, epithelial plaque like thickenings and mucous cells [3, 5]. Magnusson et al. stated that only 0.012% of the cysts in the oral cavity fulfilled the GOC criteria microscopically [7]. GOC is important to recognise due to its high recurrence rate of 30–50% [5, 6]. Most patients are treated by invasive measures such as enucleation with or without curettage and peripheral osteotomy [8], however more aggressive treatment has been recommended for larger lesions to reduce the risk of recurrence [8]. Rarely metaplastic cart
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