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Recently, genetic screening identified heterozygous\n                    <jats:italic>KCNMA1<\/jats:italic>\n                    variants in a subset of patients with debilitating paroxysmal non-kinesigenic dyskinesia, presenting with or without epilepsy (PNKD3). However, the relevance of\n                    <jats:italic>KCNMA1<\/jats:italic>\n                    mutations and the basis for clinical heterogeneity in PNKD3 has not been established. Here, we evaluate the relative severity of three\n                    <jats:italic>KCNMA1<\/jats:italic>\n                    patient variants in BK channels, neurons, and mice. In heterologous cells, BK\n                    <jats:sup>N999S<\/jats:sup>\n                    and BK\n                    <jats:sup>D434G<\/jats:sup>\n                    channels displayed gain-of-function (GOF) properties, whereas BK\n                    <jats:sup>H444Q<\/jats:sup>\n                    channels showed loss-of-function (LOF) properties. The relative degree of channel activity was BK\n                    <jats:sup>N999S<\/jats:sup>\n                    &gt; BK\n                    <jats:sup>D434G<\/jats:sup>\n                    &gt;WT &gt; BK\n                    <jats:sup>H444Q<\/jats:sup>\n                    . BK currents and action potential firing were increased, and seizure thresholds decreased, in\n                    <jats:italic>Kcnma1<\/jats:italic>\n                    <jats:sup>N999S\/WT<\/jats:sup>\n                    and\n                    <jats:italic>Kcnma1<\/jats:italic>\n                    <jats:sup>D434G\/WT<\/jats:sup>\n                    transgenic mice but not\n                    <jats:italic>Kcnma1<\/jats:italic>\n                    <jats:sup>H444Q\/WT<\/jats:sup>\n                    mice. In a novel behavioral test for paroxysmal dyskinesia, the more severely affected\n                    <jats:italic>Kcnma1<\/jats:italic>\n                    <jats:sup>N999S\/WT<\/jats:sup>\n                    mice became immobile after stress. This was abrogated by acute dextroamphetamine treatment, consistent with PNKD3-affected individuals. Homozygous\n                    <jats:italic>Kcnma1<\/jats:italic>\n                    <jats:sup>D434G\/D434G<\/jats:sup>\n                    mice showed similar immobility, but in contrast, homozygous\n                    <jats:italic>Kcnma1<\/jats:italic>\n                    <jats:sup>H444Q\/H444Q<\/jats:sup>\n                    mice displayed hyperkinetic behavior. These data establish the relative pathogenic potential of patient alleles as N999S&gt;D434G&gt;H444Q and validate\n                    <jats:italic>Kcnma1<\/jats:italic>\n                    <jats:sup>N999S\/WT<\/jats:sup>\n                    mice as a model for PNKD3 with increased seizure propensity.\n                  <\/jats:p>","DOI":"10.7554\/elife.77953","type":"journal-article","created":{"date-parts":[[2022,6,9]],"date-time":"2022-06-09T03:15:17Z","timestamp":1654744517000},"update-policy":"https:\/\/doi.org\/10.7554\/elife.77953","source":"Crossref","is-referenced-by-count":37,"title":["BK channel properties correlate with neurobehavioral severity in three KCNMA1-linked channelopathy mouse models"],"prefix":"10.7554","volume":"11","author":[{"given":"Su Mi","family":"Park","sequence":"first","affiliation":[{"name":"Department of Physiology, University of Maryland School of Medicine","place":["Baltimore, United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cooper E","family":"Roache","sequence":"additional","affiliation":[{"name":"Department of Physiology, University of Maryland School of Medicine","place":["Baltimore, United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"suffix":"II","given":"Philip H","family":"Iffland","sequence":"additional","affiliation":[{"name":"Department of Neurology, University of Maryland School of Medicine","place":["Baltimore, United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hans J","family":"Moldenhauer","sequence":"additional","affiliation":[{"name":"Department of Physiology, University of Maryland School of Medicine","place":["Baltimore, United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5094-5449","authenticated-orcid":true,"given":"Katia K","family":"Matychak","sequence":"additional","affiliation":[{"name":"Department of Physiology, University of Maryland School of Medicine","place":["Baltimore, United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amber E","family":"Plante","sequence":"additional","affiliation":[{"name":"Department of Physiology, University of Maryland School of Medicine","place":["Baltimore, United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Abby G","family":"Lieberman","sequence":"additional","affiliation":[{"name":"Department of Pharmacology, University of Maryland School of Medicine","place":["Baltimore, United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peter B","family":"Crino","sequence":"additional","affiliation":[{"name":"Department of Neurology, University of Maryland School of Medicine","place":["Baltimore, United 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