AI Article Synopsis

  • * Novel sequence variants (c.1168C>G and c.2209_2210del) were identified as potential causes of severe axonal neuropathy, hearing loss, and other symptoms in two siblings.
  • * The pathogenicity of these mutations is supported by genetic analysis and evolutionary studies, suggesting SBF1 mutations can lead to a new form of autosomal recessive axonal neuropathy (AR-CMT2) alongside CMT4B3.

Article Abstract

Biallelic mutations in the SBF1 gene have been identified in one family with demyelinating Charcot-Marie-Tooth disease (CMT4B3) and two families with axonal neuropathy and additional neurological and skeletal features. Here we describe novel sequence variants in SBF1 (c.1168C>G and c.2209_2210del) as the potential causative mutations in two siblings with severe axonal neuropathy, hearing loss, facial weakness and bulbar features. Pathogenicity of these variants is supported by co-segregation and in silico analyses and evolutionary conservation. Our findings suggest that SBF1 mutations may cause a syndromic form of autosomal recessive axonal neuropathy (AR-CMT2) in addition to CMT4B3.

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http://dx.doi.org/10.1007/s10048-016-0505-1DOI Listing

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