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DOI 10.1007/s11011-022-01079-1

Медицинская библиотека · Metabolic brain disease · 2022

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

Cianci Vittoria, Pascarella Angelo, Gasparini Sara, Donadio Vincenzo, Liguori Rocco, Incensi Alex, Rao Carmelo Massimiliano, Franzutti Claudio, Scappatura Giuseppe, Aguglia Umberto, Ferlazzo Edoardo

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Аннотация

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.

Для цитирования: Cianci Vittoria, Pascarella Angelo, Gasparini Sara, Donadio Vincenzo, Liguori Rocco, Incensi Alex, Rao Carmelo Massimiliano, Franzutti Claudio, Scappatura Giuseppe, Aguglia Umberto, Ferlazzo Edoardo. Late-onset Fabry disease due to a new (p.Pro380Leu) pathogenic variant of GLA Gene.. Metabolic brain disease. 2022. DOI: 10.1007/s11011-022-01079-1.

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