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@ARTICLE{Chivukula:878374,
author = {Chivukula, Aparna Sharma and Suslova, Mariia and Kortzak,
Daniel and Kovermann, Peter and Fahlke, Christoph},
title = {{F}unctional consequences of {SLC}1{A}3 mutations
associated with episodic ataxia 6},
journal = {Human mutation},
volume = {41},
number = {11},
issn = {1098-1004},
address = {New York, NY [u.a.]},
publisher = {Wiley-Liss},
reportid = {FZJ-2020-02814},
pages = {1892-1905},
year = {2020},
abstract = {The episodic ataxias (EA) are a group of inherited
neurological diseases characterized by paroxysmal cerebellar
incoordination. There exist nine forms of episodic ataxia
with distinct neurological symptoms and genetic origins.
Episodic ataxia type 6 (EA6) differs from other EA forms in
long attack duration, epilepsy and absent myokymia,
nystagmus, and tinnitus. It has been described in seven
families, and mutations in SLC1A3 , the gene encoding the
glial glutamate transporter EAAT1, were reported in each
family. How these mutations affect EAAT1 expression,
subcellular localization and function and how such
alterations result in the complex neurological phenotype of
EA6 is insufficiently understood. We here compare the
functional consequences of all currently known mutations by
heterologous expression in mammalian cells, biochemistry,
confocal imaging and whole‐cell patch clamp recordings of
EAAT1 transport and anion currents. We observed impairments
of multiple EAAT1 properties ranging from changes in
transport function, impaired trafficking to increased
protein expression. Many mutations caused only slight
changes illustrating how sensitively the cerebellum reacts
on impaired EAAT1 functions.},
cin = {IBI-1},
ddc = {610},
cid = {I:(DE-Juel1)IBI-1-20200312},
pnm = {553 - Physical Basis of Diseases (POF3-553)},
pid = {G:(DE-HGF)POF3-553},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:32741053},
UT = {WOS:000567441900001},
doi = {10.1002/humu.24089},
url = {https://juser.fz-juelich.de/record/878374},
}