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下颌骨后部单囊性促结缔组织增生性成釉细胞瘤:极其罕见病例报告及文献复习

Unicystic desmoplastic osteo-ameloblastoma of posterior mandible: Report of rarest of the rare case and review of literature.

作者信息

Shah Setu P, Mehta Deval, Madan Sonal, Gandhi Binita

机构信息

Department of Oral and Maxillofacial Surgery, College of Dental Sciences and Research Centre, Manipur, Ahmedabad, Gujarat, India.

Department of Oral and Maxillofacial Pathology, College of Dental Sciences and Research Centre, Manipur, Ahmedabad, Gujarat, India.

出版信息

J Oral Maxillofac Pathol. 2022 Oct-Dec;26(4):583-588. doi: 10.4103/jomfp.jomfp_20_22. Epub 2022 Dec 22.

Abstract

Desmoplastic ameloblastoma (DA) is an epithelial odontogenic tumor and a rare variant of ameloblastoma. It was first described by Eversole in 1984. In the World Health Organization (WHO) classification of odontogenic tumors (2005), DA has been considered as a distinct entity from conventional/multicystic ameloblastoma. DA differs strikingly in its clinical, radiological, and histopathological presentation when compared to other variants of ameloblastoma. We report here an extremely rare "Hybrid DA" in a 50-year-old female patient with painless hard swelling involving right posterior mandible with detailed clinical history, an unusual radiographic and histopathological presentation. Histopathology revealed odontogenic epithelium in the form of follicles, ameloblastoma with cystic degeneration, and squamous metaplasia at places and elsewhere there were odosntogenic islands compressed by dense fibrocellular stroma suggestive of desmoplasia along with osseous tissue formation. Also review of the literature and possible explanation of etiopathogenesis of cystic change and osseous tissue formation in DA are discussed.

摘要

促结缔组织增生性成釉细胞瘤(DA)是一种上皮性牙源性肿瘤,是成釉细胞瘤的一种罕见变异型。它于1984年由埃弗索尔首次描述。在世界卫生组织(WHO)牙源性肿瘤分类(2005年)中,DA被认为是与传统/多囊性成釉细胞瘤不同的实体。与成釉细胞瘤的其他变异型相比,DA在临床、放射学和组织病理学表现上有显著差异。我们在此报告一例极为罕见的“混合型DA”,发生于一名50岁女性患者,右侧下颌后部出现无痛性硬性肿胀,并伴有详细的临床病史、不寻常的影像学和组织病理学表现。组织病理学显示,病变中有呈滤泡状的牙源性上皮、伴有囊性变的成釉细胞瘤,局部有鳞状化生,其他部位有被致密纤维细胞性间质压迫的牙源性岛,提示有促结缔组织增生,同时伴有骨组织形成。此外,还讨论了文献复习以及对DA中囊性变和骨组织形成的病因学可能解释。

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