Odontogenic Choriostoma in buccal mucosa associated with suspected Goldenhar Syndrome

P. Battepati, Priyanka Gupta, B. Manjunatha, B. S. Manasali
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Abstract

Presence of teeth or tooth like masses at unusual sites of perioral structures like maxillary sinus, nasal cavity, ear, lip etc have been occasionally reported. But presence of fully developed dental structures with supporting skeletal tissue at non tooth bearing areas is extremely rare entity. In this report a similar case of presence of supplemental dental tissue along with supporting skeletal structure at buccal mucosa affecting facial symmetry which was associated with suspected Goldenhar Syndrome is presented. Report of such condition is extremely scanty in available literature and has been termed as OdotogenicChoriostoma by certain authors. *Correspondence to: Manjunatha BS, Associate Professor, Department of Oral Biology, Chairman, Scientific Presentation Committee, Member, Examination Unit, Faculty of Dentistry, Taif University, Kingdom of Saudi Arabia, E-mail: drmanju26@hotmail.com Received: April 09, 2019; Accepted: April 25, 2019; Published: April 29, 2019 Background Presence of tooth or tooth like structures at extragingival sites is not uncommon. Various extra gingival sites are reported for presence of such kind of structures like nasal cavity, maxillary sinus, ear, tongue as well as mandibular condylar and coronoid process. Presence of such structures may cause malocclusion, difficulty in breathing, incomplete articulation and speech alteration. Some of them are diagnosed early whereas some remain subclinical and diagnosed incidentally on radiograph. Confirmatory diagnosis for such structures is made by co relating clinical, radiographic and histological examination. Presence of tooth like structures at unusual site is considered ectopic and their presence in soft tissue is seldom observed. The term ‘‘choriostoma’’ has been proposed for such kind of structures which are histologically normal for a part of body except at the site where it is located [1]. Primary classification of choriostoma has been proposed based on its clinical and histomorphologic features like salivary gland choriostoma, osseous and cartilaginous choriostoma, glial and gastric choriostoma etc [2]. Osseous and cartilageous types of choriostoma are most commonly seen in oral cavity [1]. For presentation of such lesions with dental component as well as its location on extra gingival site, term ‘odontogenic choriostoma’ seems to be appropriate. We present an unusual case of an 8 year old girl with presence of tooth like structures in right buccal mucosa with supporting bony extension from right maxillary posterior region associated with suspected case of Goldenhar syndrome, for which term odontogenic choriostoma is used.
口腔黏膜牙源性绒毛膜瘤与疑似Goldenhar综合征相关
在口腔周围结构的异常部位,如上颌窦、鼻腔、耳、唇等,偶尔有牙齿或牙样肿块的报道。但是,在非牙齿承载区域具有完全发育的牙齿结构和支持骨骼组织的存在是极其罕见的实体。在本报告中,提出了一个类似的病例,在口腔黏膜处存在辅助牙组织和支撑骨骼结构,影响面部对称,这与疑似Goldenhar综合征有关。在现有文献中,这种情况的报道极其稀少,某些作者将其称为耳源性绒毛膜瘤。*通讯对象:Manjunatha BS,沙特阿拉伯塔伊夫大学口腔生物学系副教授,科学报告委员会主席,牙科学院考试组成员,E-mail: drmanju26@hotmail.com录用日期:2019年4月25日;在龈外部位出现牙齿或类似牙齿的结构并不罕见。据报道,在不同的牙龈外部位存在这类结构,如鼻腔、上颌窦、耳、舌以及下颌髁突和冠突。这些结构的存在可能导致错颌、呼吸困难、发音不全和言语改变。其中一些是早期诊断的,而另一些则是亚临床的,在x光片上偶然诊断出来的。这种结构的确诊是通过相关的临床、放射学和组织学检查做出的。在不寻常的部位出现牙齿样结构被认为是异位的,它们在软组织中的存在是很少观察到的。“绒毛膜瘤”一词已被提出用于这种结构,这种结构在组织学上对身体的一部分是正常的,除了它所在的部位。绒毛膜瘤的主要分类是根据其临床和组织形态学特征,如唾液腺绒毛膜瘤、骨性和软骨性绒毛膜瘤、胶质性和胃绒毛膜瘤等。骨性和软骨型绒毛膜瘤最常见于口腔b[1]。对于这种带有牙齿成分的病变,以及它在牙龈外的位置,术语“牙源性绒毛膜瘤”似乎是合适的。我们报告了一个不寻常的病例,一名8岁的女孩,右颊粘膜出现齿状结构,并支持右上颌后区骨延伸,疑似Goldenhar综合征,其术语为牙源性绒毛膜瘤。
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