| Literature DB >> 36178639 |
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.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