| Literature DB >> 28975462 |
Paula Sancho1,2, Ana Sánchez-Monteagudo1,2, Antonio Collado1,2, Clara Marco-Marín3,4, Cristina Domínguez-González5, Ana Camacho6,7, Erwin Knecht2,4,8, Carmen Espinós9,10,11, Vincenzo Lupo1,2,12.
Abstract
In two siblings, who suffer from an early childhood-onset axonal polyneuropathy with exclusive involvement of motor fibers, the c.629T>C (p.F210S) mutation was identified in the X-linked AIFM1 gene, which encodes for the apoptosis-inducing factor (AIF). The mutation was predicted as deleterious, according to in silico analysis. A decreased expression of the AIF protein, altered cellular morphology, and a fragmented mitochondrial network were observed in the proband's fibroblasts. This new form of motor neuropathy expands the phenotypic spectrum of AIFM1 mutations and therefore, the AIFM1 gene should be considered in the diagnosis of hereditary motor neuropathies.Entities:
Keywords: AIFM1; Apoptosis inducing factor; Gene panel; Inherited peripheral neuropathies; Mitochondrial disorder
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Year: 2017 PMID: 28975462 DOI: 10.1007/s10048-017-0524-6
Source DB: PubMed Journal: Neurogenetics ISSN: 1364-6745 Impact factor: 2.660