Suppr超能文献

一名小男孩出现吞咽困难、皮肤水疱和指甲缺失的罕见病例。

Rare case of dysphagia, skin blistering, missing nails in a young boy.

作者信息

Makker Jasbir, Bajantri Bharat, Remy Prospere

机构信息

Jasbir Makker, Bharat Bajantri, Prospere Remy, Division of Gastroenterology, Department of Medicine, Bronx Lebanon Hospital Center, Albert Einstein College of Medicine of Yeshiva University, New York, NY 10457, United States.

出版信息

World J Gastrointest Endosc. 2015 Feb 16;7(2):154-8. doi: 10.4253/wjge.v7.i2.154.

Abstract

Epidermolysis bullosa is a group of genetic disorders with an autosomal dominant or an autosomal recessive mode of inheritance and more than 300 mutations. The disorder is characterized by blistering mucocutaneous lesions and has several varying phenotypes due to anchoring defect between the epidermis and dermis. The variation in phenotypic expression depends on the involved structural protein that mediates cell adherence between different layers of the skin. Epidermolysis bullosa can also involve extra-cutaneous sites including eye, nose, ear, upper airway, genitourinary tract and gastrointestinal tract. The most prominent feature of the gastrointestinal tract involvement is development of esophageal stricture. The stricture results from recurrent esophageal mucosal blistering with consequent scarring and most commonly involves the upper esophagus. Here we present a case of a young boy with dominant subtype of dystrophic epidermolysis bullosa who presented with dysphagia, extensive skin blistering and missing nails. Management of an esophageal stricture eventually requires dilatation of the stricture or placement of a gastrostomy tube to keep up with the nutritional requirements. Gastrostomy tube also provides access for esophageal stricture dilatation in cases where antegrade approach through the mouth has failed.

摘要

大疱性表皮松解症是一组具有常染色体显性或隐性遗传模式且有300多种突变的遗传性疾病。该疾病的特征是黏膜皮肤出现水疱性病变,由于表皮与真皮之间的锚定缺陷而具有多种不同的表型。表型表达的差异取决于介导皮肤不同层之间细胞黏附的相关结构蛋白。大疱性表皮松解症还可累及皮肤外部位,包括眼、鼻、耳、上呼吸道、泌尿生殖道和胃肠道。胃肠道受累最突出的特征是食管狭窄的形成。这种狭窄是由于食管黏膜反复出现水疱并随之形成瘢痕所致,最常累及食管上段。在此,我们报告一例患有显性营养不良型大疱性表皮松解症的小男孩病例,该患儿出现吞咽困难、广泛的皮肤水疱和指甲缺失。食管狭窄的治疗最终需要对狭窄部位进行扩张或放置胃造瘘管以满足营养需求。在经口顺行途径失败的情况下,胃造瘘管还可为食管狭窄扩张提供通道。

相似文献

7
Esophageal strictures in children with recessive dystrophic epidermolysis bullosa: experience of balloon dilatation in nine cases.
J Pediatr Gastroenterol Nutr. 1998 Nov;27(5):524-9. doi: 10.1097/00005176-199811000-00006.

引用本文的文献

1
Esophageal Manifestations of Dermatological Diseases, Diagnosis and Management.皮肤病的食管表现、诊断与管理
Curr Treat Options Gastroenterol. 2022 Dec;20(4):513-528. doi: 10.1007/s11938-022-00399-6. Epub 2022 Oct 18.

本文引用的文献

3
Inherited epidermolysis bullosa.遗传性大疱性表皮松解症。
Orphanet J Rare Dis. 2010 May 28;5:12. doi: 10.1186/1750-1172-5-12.
4
Epidermolysis bullosa in Australia and New Zealand.澳大利亚和新西兰的大疱性表皮松解症。
Dermatol Clin. 2010 Apr;28(2):433-8, xvi. doi: 10.1016/j.det.2010.02.009.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验