| Literature DB >> 27982524 |
Giuseppe Piscosquito1, Stefania Magri2, Paola Saveri1, Micaela Milani2, Claudia Ciano3, Laura Farina4, Franco Taroni2, Davide Pareyson1.
Abstract
Charcot-Marie-Tooth disease type 4D (CMT4D), also known as hereditary motor and sensory neuropathy Lom type (HMSNL), is an autosomal recessive, early onset, severe demyelinating neuropathy with hearing loss, caused by N-Myc downstream-regulated gene 1 (NDRG1) mutations. CMT4D is rare with only three known mutations, one of which (p.Arg148Ter) is found in patients of Romani ancestry and accounts for the vast majority of cases. We report a 38-year-old Italian female with motor development delay, progressive neuropathy, and sensorineural deafness. Magnetic resonance imaging showed slight atrophy of cerebellum, medulla oblongata, and upper cervical spinal cord. She had a novel homozygous NDRG1 frameshift mutation (c.739delC; p.His247ThrfsTer74). The identification of this NDRG1 mutation confirms that CMT4D is not a private Romani disease and should be considered in the differential diagnosis of recessive demyelinating CMT.Entities:
Keywords: CMT4; Charcot-Marie-Tooth disease; N-Myc downstream-regulated gene 1 (NDRG1); demyelinating recessive CMT; hereditary motor and sensory neuropathy Lom type
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Year: 2017 PMID: 27982524 DOI: 10.1111/jns.12201
Source DB: PubMed Journal: J Peripher Nerv Syst ISSN: 1085-9489 Impact factor: 3.494