Literature DB >> 26725305

A splicing mutation of proteolipid protein 1 in Pelizaeus-Merzbacher disease.

Taku Omata1, Jun-Ichi Nagai2, Hiroko Shimbo3, Shiro Koizume4, Yohei Miyagi5, Kenji Kurosawa6, Sumimasa Yamashita3, Hitoshi Osaka7, Ken Inoue8.   

Abstract

A patient with an unusually mild form of Pelizaeus-Merzbacher disease was studied. Clinically, mild developmental delay with acquisition of assisted walking at 16months and mild spastic tetraplegia were evident, but no nystagmus, cerebellar, or extra-pyramidal signs were present. PLP1 mutation analysis revealed a nucleotide substitution adjacent to the acceptor site of intron 3, NM_000533.4:c.454-9T>G. Expression analysis using the patient's leukocytes demonstrated an additional abnormal transcript including the last 118bp of intron 3. In silico prediction analysis suggested the reduction of wild-type acceptor activity, which presumably evokes the cryptic splicing variant. Putative cryptic transcript results in premature termination, which may explain the mild clinical phenotype observed in this patient.
Copyright © 2015 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.

Entities:  

Keywords:  DM20; Hypomyelinating leukodystrophy; Intronic mutation; PLP1

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Substances:

Year:  2015        PMID: 26725305     DOI: 10.1016/j.braindev.2015.12.002

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


  2 in total

1.  Elucidation of the pathogenic mechanism and potential treatment strategy for a female patient with spastic paraplegia derived from a single-nucleotide deletion in PLP1.

Authors:  Keiko Yamamoto-Shimojima; Taichi Imaizumi; Yusuke Aoki; Ken Inoue; Tadashi Kaname; Yusuke Okuno; Hideki Muramatsu; Kohji Kato; Toshiyuki Yamamoto
Journal:  J Hum Genet       Date:  2019-04-19       Impact factor: 3.172

2.  Expanding the Clinical and Mutational Spectrum of the PLP1-Related Hypomyelination of Early Myelinated Structures (HEMS).

Authors:  Francesco Nicita; Chiara Aiello; Gessica Vasco; Massimiliano Valeriani; Fabrizia Stregapede; Andrea Sancesario; Michela Armando; Enrico Bertini
Journal:  Brain Sci       Date:  2021-01-13
  2 in total

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