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羊膜带综合征:两例独特临床表现的报告。

Amniotic constriction band: a report of two cases with unique clinical presentations.

作者信息

Richardson Sunil, Khandeparker Rakshit Vijay, Pellerin Philippe

机构信息

Richardsons Dental and Craniofacial Hospital, Nagercoil, India.

Department of National Reference, Center for Rare Craniomaxillofacial Malformations, Centre Hospitalier Régional Universitaire de Lille, Lille, France.

出版信息

J Korean Assoc Oral Maxillofac Surg. 2017 Jun;43(3):171-177. doi: 10.5125/jkaoms.2017.43.3.171. Epub 2017 Jun 28.

Abstract

Amniotic constriction band is a rare clinical entity with varied manifestations that range from a combination of congenital malformations to isolated malformations that are unique to each patient. The etiology of this entity remains unknown. Herein, we highlight two cases of amniotic constriction band that presented to our unit with unique clinical characteristics. To the best of our knowledge, an isolated circumferential band of scarring on the face with ocular involvement, as demonstrated by the first case, and a combination of bilateral complete cleft lip and palate with bilateral microphthalmia, auto-amputation of the right thumb, and a constriction band on the left thumb, as demonstrated by the second case, are extremely rare presentations of amniotic constriction band that were not previously reported in the literature and therefore necessitate a special mention. We discuss potential etiologies for these cases and review the existing literature on this entity.

摘要

羊膜带综合征是一种罕见的临床病症,表现多样,从先天性畸形组合到每个患者独有的孤立畸形。该病症的病因尚不清楚。在此,我们重点介绍两例具有独特临床特征的羊膜带综合征病例。据我们所知,如首例所示,面部出现孤立的环形瘢痕带并累及眼部,以及如第二例所示,双侧完全性唇腭裂合并双侧小眼畸形、右手拇指自截和左手拇指有束带,这些都是羊膜带综合征极为罕见的表现,此前文献中未见报道,因此有必要特别提及。我们讨论了这些病例的潜在病因,并回顾了关于该病症的现有文献。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/849c/5529191/210b4c3a0ecd/jkaoms-43-171-g001.jpg

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