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先天性弹性过度综合征(Ehlers-Danlos 综合征)的自然病史和表现:21 例患者的初步研究。

Natural history and manifestations of the hypermobility type Ehlers-Danlos syndrome: a pilot study on 21 patients.

机构信息

Medical Genetics, Department of Experimental Medicine, Sapienza University, San Camillo-Forlanini Hospital, Rome, Italy.

出版信息

Am J Med Genet A. 2010 Mar;152A(3):556-64. doi: 10.1002/ajmg.a.33231.

Abstract

Hypermobility type Ehlers-Danlos syndrome (HT-EDS) is a relatively frequent, although commonly misdiagnosed variant of Ehlers-Danlos syndrome, mainly characterized by marked joint instability and mild cutaneous involvement. Chronic pain, asthenia, and gastrointestinal and pelvic dysfunction are characteristic additional manifestations. We report on 21 HT-EDS patients selected from a group of 40 subjects with suspected mild hereditary connective tissue disorder. General, mucocutaneous, musculoskeletal, cardiovascular, neurologic, gastrointestinal, urogynecological, and ear-nose-throat abnormalities are investigated systematically and tabulated. Six distinct clinical presentations of HT-EDS are outlined, whose tabulation is a mnemonic for the practicing clinical geneticist in an attempt to diagnose this condition accurately. With detailed clinical records and phenotype comparison among patients of different ages, the natural history of the disorder is defined. Three phases (namely, hypermobility, pain, and stiffness) are delineated based on distinguishing manifestations. A constellation of additional, apparently uncommon abnormalities is also identified, including dolichocolon, dysphonia, and Arnold-Chiari type I malformation. Their further investigation may contribute to an understanding of the pathogenesis of the protean manifestations of HT-EDS, and a more effective approach to the evaluation and management of affected individuals.

摘要

先天性弹性过度综合征(hypermobility type Ehlers-Danlos syndrome,HT-EDS)是一种较为常见的、但常被误诊的先天性弹性过度综合征变异型,主要表现为明显的关节不稳定和轻度皮肤受累。慢性疼痛、乏力以及胃肠道和盆腔功能障碍是其特征性的附加表现。我们从一组 40 名疑似轻度遗传性结缔组织疾病患者中选择了 21 名 HT-EDS 患者进行报告。系统地调查和列表记录了一般、黏膜皮肤、肌肉骨骼、心血管、神经、胃肠道、泌尿妇科和耳鼻喉异常。概述了 HT-EDS 的 6 种不同临床表现,其列表是临床遗传学家的记忆辅助工具,旨在准确诊断这种疾病。通过详细的临床记录和不同年龄患者的表型比较,确定了该疾病的自然病史。根据不同的表现,将其分为三个阶段(即过度活动期、疼痛期和僵硬期)。还确定了一组其他的、看似不常见的异常,包括长结肠、发音困难和 Arnold-Chiari Ⅰ型畸形。对这些异常的进一步研究可能有助于理解 HT-EDS 多变表现的发病机制,并为评估和管理受影响个体提供更有效的方法。

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