| Literature DB >> 26725305 |
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.Entities:
Keywords: DM20; Hypomyelinating leukodystrophy; Intronic mutation; PLP1
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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