Bardet-Biedl综合征:目前的观点和临床前景。

IF 2.3 3区 医学 Q2 HEALTH CARE SCIENCES & SERVICES
Andrea Melluso, Floriana Secondulfo, Giovanna Capolongo, Giovambattista Capasso, Miriam Zacchia
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引用次数: 4

摘要

Bardet Biedl综合征(BBS)是一种罕见的遗传性疾病,被认为是一种非运动性纤毛病的模型。事实上,它是由编码主要位于纤毛基部的蛋白质的基因突变引起的。BBS患者的临床特征与其他纤毛病患者,特别是常染色体隐性综合征患者广泛共享;此外,纤毛相关基因的突变可引起不同的临床纤毛病实体。除了最著名的临床特征,如视网膜变性、学习障碍、多指畸形、肥胖和肾脏缺陷,BBS中还报道了一些其他的临床症状,扩大了我们对其临床谱复杂性的理解。本综述旨在描述目前对BBS的认识i)病理生理学,ii)临床表现,强调最常见和较少描述的特征,iii)当前和未来的治疗前景。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Bardet-Biedl Syndrome: Current Perspectives and Clinical Outlook.

Bardet-Biedl Syndrome: Current Perspectives and Clinical Outlook.

Bardet-Biedl Syndrome: Current Perspectives and Clinical Outlook.

The Bardet Biedl syndrome (BBS) is a rare inherited disorder considered a model of non-motile ciliopathy. It is in fact caused by mutations of genes encoding for proteins mainly localized to the base of the cilium. Clinical features of BBS patients are widely shared with patients suffering from other ciliopathies, especially autosomal recessive syndromic disorders; moreover, mutations in cilia-related genes can cause different clinical ciliopathy entities. Besides the best-known clinical features, as retinal degeneration, learning disabilities, polydactyly, obesity and renal defects, several additional clinical signs have been reported in BBS, expanding our understanding of the complexity of its clinical spectrum. The present review aims to describe the current knowledge of BBS i) pathophysiology, ii) clinical manifestations, highlighting both the most common and the less described features, iii) current and future perspective for treatment.

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来源期刊
Therapeutics and Clinical Risk Management
Therapeutics and Clinical Risk Management HEALTH CARE SCIENCES & SERVICES-
CiteScore
4.80
自引率
3.60%
发文量
139
审稿时长
16 weeks
期刊介绍: Therapeutics and Clinical Risk Management is an international, peer-reviewed journal of clinical therapeutics and risk management, focusing on concise rapid reporting of clinical studies in all therapeutic areas, outcomes, safety, and programs for the effective, safe, and sustained use of medicines, therapeutic and surgical interventions in all clinical areas. The journal welcomes submissions covering original research, clinical and epidemiological studies, reviews, guidelines, expert opinion and commentary. The journal will consider case reports but only if they make a valuable and original contribution to the literature. As of 18th March 2019, Therapeutics and Clinical Risk Management will no longer consider meta-analyses for publication. The journal does not accept study protocols, animal-based or cell line-based studies.
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