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一种罕见综合征中的颅颌面表现:口面指综合征IV型(莫尔-马耶夫斯基综合征)

Craniodentofacial Manifestations in a Rare Syndrome: Orofaciodigital Type IV (Mohr-Majewski Syndrome).

作者信息

Ozdemir-Karatas Meltem, Ozdemir-Ozenen Didem, Hart P Suzanne, Hart Thomas C

机构信息

Department of Maxillofacial Prosthodontics, Faculty of Dentistry, Istanbul University, Capa, Istanbul, Turkey.

Department of Pedodontics, Faculty of Dentistry, Yeditepe University, Goztepe, Istanbul, Turkey.

出版信息

Case Rep Dent. 2014;2014:605892. doi: 10.1155/2014/605892. Epub 2014 Dec 21.

Abstract

Background. The orofaciodigital syndromes (OFDS) are a heterogeneous group of syndromes that affect the face, oral cavity, and the digits. OFDS type IV (OMIM %258860) is rare and characterized by broad nasal root and tip, orbital hypertelorism or telecanthus, micrognathia, hypoplastic mandible, and low-set ears. Oral symptoms may include cleft lip, cleft or highly arched palate, bifid uvula, cleft or hypoplastic maxillary and mandibular alveolar ridge, oral frenula, lingual hamartoma, and absent or hypoplastic epiglottis. Dental anomalies are common and generally include disturbances in the number of teeth. Case Report. This report presents a six-year-old girl, referred with the chief complaint of missing teeth. She was diagnosed as having OFDS type IV based on clinical findings. Her parents reported three deceased children and two fetuses that had the same phenotype. She was the seventh child of consanguineous parents who were first cousins. Conclusion. This is a very rare syndrome. Many reported OFDS type IV cases have consanguineous parents, consistent with an autosomal recessive trait. Manifestation of cleft palate in the healthy sibling may be mild expression of the disorder or an unrelated isolated cleft.

摘要

背景。口面指综合征(OFDS)是一组影响面部、口腔和手指的异质性综合征。IV型口面指综合征(OMIM %258860)较为罕见,其特征为鼻根宽阔且鼻尖宽大、眶距增宽或内眦间距增宽、小颌畸形、下颌骨发育不全以及低位耳。口腔症状可能包括唇裂、腭裂或高拱腭、悬雍垂裂、上颌和下颌牙槽嵴裂或发育不全、口腔系带、舌错构瘤以及会厌缺如或发育不全。牙齿异常很常见,通常包括牙齿数量紊乱。病例报告。本报告介绍了一名6岁女孩,其主要诉求为牙齿缺失。根据临床检查结果,她被诊断为IV型口面指综合征。她的父母报告说有3个已故子女和2个胎儿具有相同的表型。她是近亲结婚(表亲)父母的第7个孩子。结论。这是一种非常罕见的综合征。许多报告的IV型口面指综合征病例其父母为近亲,这与常染色体隐性遗传特征相符。健康同胞中出现腭裂可能是该疾病的轻度表现,或者是一种无关的孤立性腭裂。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/71b9/4283451/03f0b3a07473/CRID2014-605892.001.jpg

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