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Spectrum of KV 2.1 Dysfunction in KCNB1-Associated Neurodevelopmental Disorders.

Seok Kyu KangCarlos G VanoyeSunita N MisraDennis M EchevarriaJeffrey D CalhounJohn B O'ConnorKatarina L FabreDianalee McKnightLaurie DemmerPaula GoldenbergLauren E GroteIsabelle ThiffaultCarol SaundersKevin A StraussAli TorkamaniJasper van der SmagtKoen van GassenRobert P CarsonJullianne DiazEyby LeonJoseph E JacherMark C HannibalJessica LitwinNeil R FriedmanAllison SchreiberBryan LynchAnnapurna PoduriEric D MarshEthan M GoldbergJohn J MillichapAlfred L GeorgeJennifer A Kearney
Published in: Annals of neurology (2019)
Our study establishes a platform for rapid screening of KV 2.1 functional defects caused by KCNB1 variants associated with DEE and other NDDs. This will aid in establishing KCNB1 variant pathogenicity and the mechanism of dysfunction, which will enable targeted strategies for therapeutic intervention based on molecular phenotype. ANN NEUROL 2019;86:899-912.
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