Literature DB >> 36178639

Late-onset Fabry disease due to a new (p.Pro380Leu) pathogenic variant of GLA Gene.

Vittoria Cianci1, Angelo Pascarella1,2, Sara Gasparini1,2, Vincenzo Donadio3, Rocco Liguori3, Alex Incensi3, Carmelo Massimiliano Rao4, Claudio Franzutti5, Giuseppe Scappatura5, Umberto Aguglia1,2,6, Edoardo Ferlazzo7,8,9.   

Abstract

Fabry disease is a rare X-linked lysosomal storage disorder due to pathogenic variants of the galactosidase alpha (GLA) gene, leading to a deficiency of alpha-galactosidase A. The inadequate enzymatic activity leads to progressive glycosphingolipids accumulation within tissues and subsequent multi-systemic dysfunction, with predominant involvement of heart, kidney, and nervous system. Two subtypes are recognized: the classic type and the late-onset type. We here describe the clinical characteristics of a patient with late-onset Fabry disease carrying a not previously identified GLA gene variant. This 50-year-old man came to hospital because of an acute ischemic stroke. He also complained of acroparesthesia and had angiokeratomas in the nape and the back. Blood alpha-galactosidase A activity was low, plasmatic lyso-Gb3 level was borderline, cardiac MRI showed cardiac fibrosis, brain MRI documented cerebrovascular disease, and skin biopsy revealed small fiber neuropathy without globotriaosylceramide-3 skin deposits. Genetic study by means of targeted next-generation sequencing analysis disclosed a missense substitution c.1139C>T (p.Pro380Leu) in the GLA gene. We suggest that this novel variant should be considered as pathogenic and associated with a late-onset variant of Fabry disease with a predominant neurological phenotype.
© 2022. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.

Entities:  

Keywords:  GLA pathogenic variant; Late-onset Fabry Disease; Small fiber neuropathy; Stroke

Year:  2022        PMID: 36178639     DOI: 10.1007/s11011-022-01079-1

Source DB:  PubMed          Journal:  Metab Brain Dis        ISSN: 0885-7490            Impact factor:   3.655


  11 in total

Review 1.  When and How to Diagnose Fabry Disease in Clinical Pratice.

Authors:  Martin Michaud; Wladimir Mauhin; Nadia Belmatoug; Roselyne Garnotel; Naiya Bedreddine; Florian Catros; Sophie Ancellin; Olivier Lidove; Francis Gaches
Journal:  Am J Med Sci       Date:  2020-07-10       Impact factor: 2.378

Review 2.  Fabry disease revisited: Management and treatment recommendations for adult patients.

Authors:  Alberto Ortiz; Dominique P Germain; Robert J Desnick; Juan Politei; Michael Mauer; Alessandro Burlina; Christine Eng; Robert J Hopkin; Dawn Laney; Aleš Linhart; Stephen Waldek; Eric Wallace; Frank Weidemann; William R Wilcox
Journal:  Mol Genet Metab       Date:  2018-02-28       Impact factor: 4.797

Review 3.  Small fiber neuropathy in Fabry disease.

Authors:  Marieke Biegstraaten; Carla E M Hollak; Mayienne Bakkers; Catharina G Faber; Johannes M F G Aerts; Ivo N van Schaik
Journal:  Mol Genet Metab       Date:  2012-03-24       Impact factor: 4.797

4.  Elevated globotriaosylsphingosine is a hallmark of Fabry disease.

Authors:  Johannes M Aerts; Johanna E Groener; Sijmen Kuiper; Wilma E Donker-Koopman; Anneke Strijland; Roelof Ottenhoff; Cindy van Roomen; Mina Mirzaian; Frits A Wijburg; Gabor E Linthorst; Anouk C Vedder; Saskia M Rombach; Josanne Cox-Brinkman; Pentti Somerharju; Rolf G Boot; Carla E Hollak; Roscoe O Brady; Ben J Poorthuis
Journal:  Proc Natl Acad Sci U S A       Date:  2008-02-19       Impact factor: 11.205

5.  Stroke in Fabry disease frequently occurs before diagnosis and in the absence of other clinical events: natural history data from the Fabry Registry.

Authors:  Katherine Sims; Juan Politei; Maryam Banikazemi; Philip Lee
Journal:  Stroke       Date:  2009-01-15       Impact factor: 7.914

6.  A comprehensive testing algorithm for the diagnosis of Fabry disease in males and females.

Authors:  Ashlee R Stiles; Haoyue Zhang; Jian Dai; Patricia McCaw; James Beasley; Catherine Rehder; Dwight D Koeberl; Marie McDonald; Deeksha S Bali; Sarah P Young
Journal:  Mol Genet Metab       Date:  2020-05-03       Impact factor: 4.797

7.  Uncertain diagnosis of fabry disease in patients with neuropathic pain, angiokeratoma or cornea verticillata: consensus on the approach to diagnosis and follow-up.

Authors:  L van der Tol; David Cassiman; Gunnar Houge; Mirian C Janssen; Robin H Lachmann; Gabor E Linthorst; Uma Ramaswami; Claudia Sommer; Camilla Tøndel; Michael L West; Frank Weidemann; Frits A Wijburg; Einar Svarstad; Carla Em Hollak; Marieke Biegstraaten
Journal:  JIMD Rep       Date:  2014-09-16

8.  Use of a rare disease registry for establishing phenotypic classification of previously unassigned GLA variants: a consensus classification system by a multispecialty Fabry disease genotype-phenotype workgroup.

Authors:  Dominique P Germain; João Paulo Oliveira; Daniel G Bichet; Han-Wook Yoo; Robert J Hopkin; Roberta Lemay; Juan Politei; Christoph Wanner; William R Wilcox; David G Warnock
Journal:  J Med Genet       Date:  2020-03-11       Impact factor: 6.318

9.  Cornea verticillata and acroparesthesia efficiently discriminate clusters of severity in Fabry disease.

Authors:  Wladimir Mauhin; Olivier Benveniste; Damien Amelin; Clémence Montagner; Foudil Lamari; Catherine Caillaud; Claire Douillard; Bertrand Dussol; Vanessa Leguy-Seguin; Pauline D'Halluin; Esther Noel; Thierry Zenone; Marie Matignon; François Maillot; Kim-Heang Ly; Gérard Besson; Marjolaine Willems; Fabien Labombarda; Agathe Masseau; Christian Lavigne; Didier Lacombe; Hélène Maillard; Olivier Lidove
Journal:  PLoS One       Date:  2020-05-22       Impact factor: 3.240

10.  Skin globotriaosylceramide 3 deposits are specific to Fabry disease with classical mutations and associated with small fibre neuropathy.

Authors:  Rocco Liguori; Alex Incensi; Silvia de Pasqua; Renzo Mignani; Enrico Fileccia; Marisa Santostefano; Elena Biagini; Claudio Rapezzi; Silvia Palmieri; Ilaria Romani; Walter Borsini; Alessandro Burlina; Roberto Bombardi; Marco Caprini; Patrizia Avoni; Vincenzo Donadio
Journal:  PLoS One       Date:  2017-07-03       Impact factor: 3.240

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