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多学科方法在两个棘手病例中的重要性:法布里病的完美匹配

The importance of a multidisciplinary approach in two tricky cases: the perfect match for Fabry disease.

作者信息

Berti Gian Marco, Aiello Valeria, Vischini Gisella, Lerario Sarah, Ciurli Francesca, Santostefano Marisa, Donadio Vincenzo, Biagini Elena, Fresina Michela, Fabbrizio Benedetta, Montanari Francesca, Turchetti Daniela, Pasquinelli Gianandrea, Mignani Renzo, La Manna Gaetano, Capelli Irene

机构信息

Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum - University of Bologna, Bologna, Italy.

Nephrology, Dialysis and Kidney Transplant Unit, IRCCS Azienda Ospedaliero-Universitaria di Bologna, Bologna, Italy.

出版信息

BMC Nephrol. 2025 Feb 13;26(1):77. doi: 10.1186/s12882-025-04009-2.

Abstract

Anderson-Fabry disease (AFD) is a multisystem X-linked lysosomal storage disorder caused by a deficiency in the enzyme α-galactosidase A (α-Gal A). This deficiency results in the intracellular accumulation of glycosphingolipids, primarily uncleaved globotriaosylceramide (Gb3) and its deacylated form, lyso-globotriaosylceramide (Lyso-Gb3), leading to progressive organ damage and functional impairment. The diagnostic evaluation for AFD involves clinical assessment and family history, supported by biochemical testing (α-Gal A enzyme activity and Lyso-Gb3 levels) and genetic analysis of the GLA gene. In cases of unexplained renal impairment or when genetic analysis is inconclusive, kidney biopsy is often required to confirm the diagnosis and guide targeted treatments. However, histological findings in kidney biopsies may sometimes be nonspecific, complicating the diagnostic process. This article aims to provide an updated perspective on the role of kidney biopsy in AFD, illustrating two cases that exemplify its pivotal role in confirming or excluding the suspected disease, proving to be both decisive and confounding in this complex clinical setting.

摘要

安德森-法布里病(AFD)是一种多系统X连锁溶酶体贮积症,由α-半乳糖苷酶A(α-Gal A)缺乏引起。这种缺乏导致糖鞘脂在细胞内蓄积,主要是未裂解的球三糖基神经酰胺(Gb3)及其脱酰基形式溶血尿苷酰鞘氨醇(Lyso-Gb3),从而导致进行性器官损害和功能障碍。AFD的诊断评估包括临床评估和家族史,并辅以生化检测(α-Gal A酶活性和Lyso-Gb3水平)以及对GLA基因的遗传分析。在不明原因的肾功能损害病例或遗传分析结果不确定时,通常需要进行肾活检以确诊并指导靶向治疗。然而,肾活检的组织学结果有时可能不具有特异性,使诊断过程复杂化。本文旨在提供关于肾活检在AFD中作用的最新观点,通过两个病例说明其在确诊或排除疑似疾病方面的关键作用,在这种复杂的临床情况下,肾活检既具有决定性作用,也会带来困惑。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e4eb/11827196/0afe51f751e0/12882_2025_4009_Fig1_HTML.jpg

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