Melluso Andrea, Secondulfo Floriana, Capolongo Giovanna, Capasso Giovambattista, Zacchia Miriam
Department of Translational Medical Sciences, University of Campania "Luigi Vanvitelli", Naples, Italy.
Biogem Scarl, Ariano Irpino, AV, 83031, Italy.
Ther Clin Risk Manag. 2023 Jan 30;19:115-132. doi: 10.2147/TCRM.S338653. eCollection 2023.
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.
巴德-比埃尔综合征(BBS)是一种罕见的遗传性疾病,被认为是非运动性纤毛病的模型。事实上,它是由主要定位于纤毛基部的蛋白质编码基因突变引起的。BBS患者的临床特征与其他纤毛病患者广泛共有,尤其是常染色体隐性综合征性疾病;此外,与纤毛相关的基因突变可导致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、肥胖和肾脏缺陷外,BBS还报告了一些其他临床体征,这扩展了我们对其临床谱复杂性的认识。本综述旨在描述BBS的以下方面的现有知识:i)病理生理学,ii)临床表现,突出最常见和较少描述的特征,iii)当前和未来的治疗前景。