Molecular Genetics & Genomic Medicine · 2018 · 110 citations · 39 references
Life SciencesGeneticsMolecular GeneticsCardiovascular GeneticsHuman GeneticsMedical GeneticsGenetic DisordersGenetic RiskInherited CardiomyopathiesMendelian Cardiovascular DisordersMendelian DisordersVariant InterpretationCardiomyopathy Genotype-phenotype CorrelationFabry DiseaseInherited VariantsPhenotypic Mutation
BACKGROUND: The p.Asn215Ser or p.N215S GLA variant has been associated with late-onset cardiac variant of Fabry disease. METHODS: To expand on the scarce phenotype data, we analyzed natural history data from 125 p.N215S patients (66 females, 59 males) enrolled in the Fabry Registry (NCT00196742) and compared it with data from 401 patients (237 females, 164 males) harboring mutations associated with classic Fabry disease. We evaluated interventricular septum thickness (IVST), left ventricular posterior wall thickness (LVPWT), estimated glomerular filtration rate and severe clinical events. RESULTS: In p.N215S males, mildly abnormal mean IVST and LVPWT values were observed in patients aged 25-34 years, and values gradually increased with advancing age. Mean values were similar to those of classic males. In p.N215S females, these abnormalities occurred primarily in patients aged 55-64 years. Severe clinical events in p.N215S patients were mainly cardiac (males 31%, females 8%) while renal and cerebrovascular events were rare. Renal impairment occurred in 17% of p.N215S males (mostly in patients aged 65-74 years), and rarely in females (3%). CONCLUSION: p.N215S is a disease-causing mutation with severe clinical manifestations found primarily in the heart. Cardiac involvement may become as severe as in classic Fabry patients, especially in males.
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Roberto M. Lang, Luigi P. Badano, Victor Mor‐Avi et al. · Journal of the American Society of Echocardiography · 2015 · 18K citations · Full text
HGVS Recommendations for the Description of Sequence Variants: 2016 Update
Johan T. den Dunnen, Raymond Dalgleish, Donna Maglott et al. · Human Mutation · 2016 · 1.7K citations · Full text
An Atypical Variant of Fabry's Disease in Men with Left Ventricular Hypertrophy
Shoichiro Nakao, Toshihiro Takenaka, Masato Maeda et al. · New England Journal of Medicine · 1995 · 765 citations · Full text
Medicine, Cardiology, Genetics +10