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@ARTICLE{Rydning:140009,
author = {Rydning, S. L. and Dudesek, A. and Rimmele, F. and Funke,
C. and Krüger, S. and Biskup, Saskia and Vigeland, M. D.
and Hjorthaug, H. S. and Sejersted, Y. and Tallaksen, C. and
Selmer, K. K. and Kamm, C.},
title = {{A} novel heterozygous variant in {ERLIN}2 causes autosomal
dominant pure hereditary spastic paraplegia.},
journal = {European journal of neurology},
volume = {25},
number = {7},
issn = {1351-5101},
address = {Oxford},
publisher = {Blackwell Science78889},
reportid = {DZNE-2020-06331},
pages = {943-e71},
year = {2018},
abstract = {Hereditary spastic paraplegias (HSPs) are clinically and
genetically heterogeneous monogenic disorders. To date,
nearly 70 genes are known to be causative. The aim of this
project was to identify the genetic cause of autosomal
dominantly inherited pure HSP in two large, unrelated
non-consanguineous families.The two families were
characterized clinically and selected members underwent
whole exome sequencing. Potentially disease-causing variants
were confirmed by Sanger sequencing and their functional
consequences on protein function were predicted by
bioinformatic prediction tools.The patients presented with
pure spastic paraplegia with age of onset between 9 and 46
years. In both families, a novel heterozygous missense
variant in ERLIN2, c.386G>C; p.Ser129Thr, was the only
potentially pathogenic variant identified that segregated
with the disease.Biallelic variants in ERLIN2 are known to
cause recessive HSP type SPG18. Here, the first two families
with an autosomal dominant, pure form of HSP caused by a
novel ERLIN2 heterozygous missense variant are described.
These findings expand the mutational and inheritance
spectrum of SPG18. ERLIN2 variants should also be considered
in the diagnostic evaluation of patients with autosomal
dominant HSP.},
keywords = {Adult / Female / Heterozygote / Humans / Male / Membrane
Proteins: genetics / Middle Aged / Mutation / Pedigree /
Spastic Paraplegia, Hereditary: genetics / ERLIN2 protein,
human (NLM Chemicals) / Membrane Proteins (NLM Chemicals)},
cin = {Rostock / Greifswald Pre 2020 / Tübingen Pre 2020},
ddc = {610},
cid = {I:(DE-2719)6000017 / I:(DE-2719)6000018},
pnm = {344 - Clinical and Health Care Research (POF3-344) / 345 -
Population Studies and Genetics (POF3-345)},
pid = {G:(DE-HGF)POF3-344 / G:(DE-HGF)POF3-345},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:29528531},
doi = {10.1111/ene.13625},
url = {https://pub.dzne.de/record/140009},
}