Literature DB >> 27982524

A novel NDRG1 mutation in a non-Romani patient with CMT4D/HMSN-Lom.

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.
© 2016 Peripheral Nerve Society.

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


  2 in total

1.  A splice altering variant in NDRG1 gene causes Charcot-Marie-Tooth disease, type 4D.

Authors:  Pooja Pravinbabu; Vikram V Holla; Prashant Phulpagar; Nitish Kamble; Manjunath Netravathi; Ravi Yadav; Pramod Kumar Pal; Babylakshmi Muthusamy
Journal:  Neurol Sci       Date:  2022-02-11       Impact factor: 3.307

2.  Aberrant Neuregulin 1/ErbB Signaling in Charcot-Marie-Tooth Type 4D Disease.

Authors:  Li-Ting Jiang; Yu-Hui Chen; Jie-Hong Huang; Wei-Fang Tong; Ling-Jing Jin; Li-Xi Li
Journal:  Mol Cell Biol       Date:  2022-06-16       Impact factor: 5.069

  2 in total

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