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Altered PLP1 splicing causes hypomyelination of...
Journal article

Altered PLP1 splicing causes hypomyelination of early myelinating structures

Abstract

OBJECTIVE: The objective of this study was to investigate the genetic etiology of the X-linked disorder "Hypomyelination of Early Myelinating Structures" (HEMS). METHODS: We included 16 patients from 10 families diagnosed with HEMS by brain MRI criteria. Exome sequencing was used to search for causal mutations. In silico analysis of effects of the mutations on splicing and RNA folding was performed. In vitro gene splicing was examined in RNA …

Authors

Kevelam SH; Taube JR; van Spaendonk RML; Bertini E; Sperle K; Tarnopolsky M; Tonduti D; Valente EM; Travaglini L; Sistermans EA

Journal

Annals of Clinical and Translational Neurology, Vol. 2, No. 6, pp. 648–661

Publisher

Wiley

Publication Date

June 2015

DOI

10.1002/acn3.203

ISSN

2328-9503