Literature DB >> 15130691

Increased NPC1 mRNA in skin fibroblasts from Niemann-Pick disease type C patients.

Toshiyuki Yamamoto1, Jian-Hua Feng, Katsumi Higaki, Miyako Taniguchi, Eiji Nanba, Haruaki Ninomiya, Kousaku Ohno.   

Abstract

Niemann-Pick disease type C (NP-C) is an autosomal recessive lipid-storage disease that is characterized by progressive neurodegeneration and hepatosplenomegaly. Since identification of the NPC1 gene in 1997, a total of 120 disease-causing mutations have been reported. In this study, two novel mutations were identified, namely c.2508[-2509]A del (837Fs-838X) in exon 16 and T3194G (V1065G) in exon 21. To explore the impact of NPC1 mutations on transcription of this gene, we analyzed NPC1 mRNA levels in skin fibroblasts derived from NP-C patients. Fibroblasts from patients with missense mutations showed increased levels of NPC1 mRNA while fibroblasts from patients with a specific frameshift mutation showed mRNA levels similar to those of normal control subjects. These results suggest that NPC1 transcription levels are altered in cells with mutations in the NPC1 gene.

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Year:  2004        PMID: 15130691     DOI: 10.1016/S0387-7604(03)00162-1

Source DB:  PubMed          Journal:  Brain Dev        ISSN: 0387-7604            Impact factor:   1.961


  2 in total

1.  Endoplasmic reticulum-associated degradation of Niemann-Pick C1: evidence for the role of heat shock proteins and identification of lysine residues that accept ubiquitin.

Authors:  Naoe Nakasone; Yuko S Nakamura; Katsumi Higaki; Nao Oumi; Kousaku Ohno; Haruaki Ninomiya
Journal:  J Biol Chem       Date:  2014-06-02       Impact factor: 5.157

2.  Modeling Niemann-Pick disease type C in a human haploid cell line allows for patient variant characterization and clinical interpretation.

Authors:  Steven Erwood; Reid A Brewer; Teija M I Bily; Eleonora Maino; Liangchi Zhou; Ronald D Cohn; Evgueni A Ivakine
Journal:  Genome Res       Date:  2019-11-21       Impact factor: 9.043

  2 in total

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