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Knockdown of the clavesin gene in zebrafish (clvs2) produced edema phenotypes due to disruption of podocyte structure and loss of glomerular filtration barrier integrity that could be rescued by WT\n                    <jats:italic>CLVS1<\/jats:italic>\n                    but not the p.H310Y variant. Analysis of cultured human podocytes with CRISPR\/Cas9-mediated\n                    <jats:italic>CLVS1<\/jats:italic>\n                    knockout or homozygous H310Y knockin revealed deficits in clathrin-mediated endocytosis and increased susceptibility to apoptosis that could be rescued with corticosteroid treatment, mimicking the steroid responsiveness observed in patients with SSNS. The p.H310Y variant also disrupted binding of clavesin-1 to\n                    <jats:bold>\u03b1<\/jats:bold>\n                    -tocopherol transfer protein, resulting in increased reactive oxygen species (ROS) accumulation in\n                    <jats:italic>CLVS1<\/jats:italic>\n                    -deficient podocytes. Treatment of\n                    <jats:italic>CLVS1<\/jats:italic>\n                    -knockout or homozygous H310Y-knockin podocytes with pharmacological ROS inhibitors restored viability to control levels. Taken together, these data identify\n                    <jats:italic>CLVS1<\/jats:italic>\n                    as a candidate gene for SSNS, provide insight into therapeutic effects of corticosteroids on podocyte cellular dynamics, and add to the growing evidence of the importance of endocytosis and oxidative stress regulation to podocyte function.\n                  <\/jats:p>","DOI":"10.1172\/jci.insight.152102","type":"journal-article","created":{"date-parts":[[2021,12,7]],"date-time":"2021-12-07T12:00:20Z","timestamp":1638878420000},"source":"Crossref","is-referenced-by-count":22,"title":["Steroid-sensitive nephrotic syndrome candidate gene CLVS1 regulates podocyte oxidative stress and endocytosis"],"prefix":"10.1172","volume":"7","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1158-1117","authenticated-orcid":true,"given":"Brandon M.","family":"Lane","sequence":"first","affiliation":[{"name":"Department of Pediatrics, Division of Nephrology, and Duke Molecular Physiology 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