Late-onset Fabry disease due to a new (p.Pro380Leu) pathogenic variant of GLA Gene.
Vittoria CianciAngelo PascarellaSara GaspariniVincenzo DonadioRocco LiguoriAlex IncensiCarmelo Massimiliano RaoClaudio FranzuttiGiuseppe ScappaturaUmberto AgugliaEdoardo FerlazzoPublished in: Metabolic brain disease (2022)
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.
Keyphrases
- late onset
- early onset
- copy number
- genome wide
- acute ischemic stroke
- replacement therapy
- heart failure
- gene expression
- healthcare
- emergency department
- oxidative stress
- hypertrophic cardiomyopathy
- left ventricular
- genome wide identification
- contrast enhanced
- magnetic resonance
- computed tomography
- atrial fibrillation
- nitric oxide
- autism spectrum disorder
- dna methylation
- blood brain barrier
- intellectual disability
- wound healing
- white matter
- soft tissue
- anti inflammatory
- transcription factor
- liver fibrosis
- cerebral ischemia
- data analysis