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@ARTICLE{Park:140830,
author = {Park, Joohyun and Koko, Mahmoud and Hedrich, Ulrike B S and
Hermann, Andreas and Cremer, Kirsten and Haberlandt, Edda
and Grimmel, Mona and Alhaddad, Bader and Beck-Woedl,
Stefanie and Harrer, Merle and Karall, Daniela and
Kingelhoefer, Lisa and Tzschach, Andreas and Matthies, Lars
C and Strom, Tim M and Ringelstein, Erich Bernd and Sturm,
Marc and Engels, Hartmut and Wolff, Markus and Lerche,
Holger and Haack, Tobias B},
title = {{KCNC}1-related disorders: new de novo variants expand the
phenotypic spectrum.},
journal = {Annals of Clinical and Translational Neurology},
volume = {6},
number = {7},
issn = {2328-9503},
address = {Chichester [u.a.]},
publisher = {Wiley},
reportid = {DZNE-2020-07152},
pages = {1319-1326},
year = {2019},
abstract = {A recurrent de novo missense variant in KCNC1, encoding a
voltage-gated potassium channel expressed in inhibitory
neurons, causes progressive myoclonus epilepsy and ataxia,
and a nonsense variant is associated with intellectual
disability. We identified three new de novo missense
variants in KCNC1 in five unrelated individuals causing
different phenotypes featuring either isolated
nonprogressive myoclonus (p.Cys208Tyr), intellectual
disability (p.Thr399Met), or epilepsy with myoclonic,
absence and generalized tonic-clonic seizures, ataxia, and
developmental delay (p.Ala421Val, three patients).
Functional analyses demonstrated no measurable currents for
all identified variants and dominant-negative effects for
p.Thr399Met and p.Ala421Val predicting neuronal
disinhibition as the underlying disease mechanism.},
keywords = {Animals / Ataxia: genetics / Child / Codon, Nonsense /
Genetic Association Studies / Humans / Intellectual
Disability: genetics / Male / Mutation, Missense / Myoclonic
Epilepsies, Progressive / Seizures: genetics / Shaw
Potassium Channels: genetics / Shaw Potassium Channels:
physiology / Xenopus laevis},
cin = {AG Teipel / Dresden common},
ddc = {610},
cid = {I:(DE-2719)1510100 / I:(DE-2719)6000013},
pnm = {344 - Clinical and Health Care Research (POF3-344)},
pid = {G:(DE-HGF)POF3-344},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:31353862},
pmc = {pmc:PMC6649617},
doi = {10.1002/acn3.50799},
url = {https://pub.dzne.de/record/140830},
}