{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T20:41:06Z","timestamp":1790800866238,"version":"4.1.0"},"reference-count":33,"publisher":"Elsevier BV","issue":"5","license":[{"start":{"date-parts":[[2023,5,1]],"date-time":"2023-05-01T00:00:00Z","timestamp":1682899200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2023,5,1]],"date-time":"2023-05-01T00:00:00Z","timestamp":1682899200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2023,4,12]],"date-time":"2023-04-12T00:00:00Z","timestamp":1681257600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["F31HL149131"],"award-info":[{"award-number":["F31HL149131"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["R35 HL144980"],"award-info":[{"award-number":["R35 HL144980"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000050","name":"National Heart, Lung, and Blood Institute","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000050","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000871","name":"Mayo Clinic","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000871","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100001316","name":"Abbott Laboratories","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100001316","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100004374","name":"Medtronic","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100004374","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001674","name":"Fondation Leducq","doi-asserted-by":"publisher","award":["18CVD05"],"award-info":[{"award-number":["18CVD05"]}],"id":[{"id":"10.13039\/501100001674","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["cell.com","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Stem Cell Reports"],"published-print":{"date-parts":[[2023,5]]},"DOI":"10.1016\/j.stemcr.2023.04.005","type":"journal-article","created":{"date-parts":[[2023,5,9]],"date-time":"2023-05-09T16:12:26Z","timestamp":1683648746000},"page":"1075-1089","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":11,"title":["Cellular and electrophysiological characterization of triadin knockout syndrome using induced pluripotent stem cell-derived cardiomyocytes"],"prefix":"10.1016","volume":"18","author":[{"given":"Daniel J.","family":"Clemens","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dan","family":"Ye","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lili","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C.S. John","family":"Kim","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wei","family":"Zhou","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Steven M.","family":"Dotzler","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David J.","family":"Tester","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Isabelle","family":"Marty","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bjorn C.","family":"Knollmann","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael J.","family":"Ackerman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.stemcr.2023.04.005_bib1","doi-asserted-by":"crossref","first-page":"2051","DOI":"10.1161\/CIRCULATIONAHA.115.015397","article-title":"Homozygous\/compound heterozygous triadin mutations associated with autosomal recessive long QT syndrome and pediatric sudden cardiac arrest: elucidation of triadin knockout syndrome","volume":"131","author":"Altmann","year":"2015","journal-title":"Circulation"},{"key":"10.1016\/j.stemcr.2023.04.005_bib2","doi-asserted-by":"crossref","first-page":"531","DOI":"10.1038\/nrcardio.2015.61","article-title":"Gene therapy to treat cardiac arrhythmias","volume":"12","author":"Bongianino","year":"2015","journal-title":"Nat. Rev. Cardiol."},{"key":"10.1016\/j.stemcr.2023.04.005_bib3","doi-asserted-by":"crossref","first-page":"340","DOI":"10.1093\/ehjci\/eux158.222","article-title":"A novel Triadin variant causes a severe clinical CPVT phenotype in two young brothers","volume":"19","author":"Broendberg","year":"2017","journal-title":"EP Europace"},{"key":"10.1016\/j.stemcr.2023.04.005_bib4","doi-asserted-by":"crossref","first-page":"855","DOI":"10.1038\/nmeth.2999","article-title":"Chemically defined generation of human cardiomyocytes","volume":"11","author":"Burridge","year":"2014","journal-title":"Nat. Methods"},{"key":"10.1016\/j.stemcr.2023.04.005_bib5","doi-asserted-by":"crossref","first-page":"171","DOI":"10.1016\/j.ymthe.2019.09.012","article-title":"Interplay between triadin and calsequestrin in the pathogenesis of CPVT in the mouse","volume":"28","author":"Cacheux","year":"2020","journal-title":"Mol. Ther."},{"key":"10.1016\/j.stemcr.2023.04.005_bib6","doi-asserted-by":"crossref","first-page":"187","DOI":"10.1093\/cvr\/cvt023","article-title":"Triadin regulates cardiac muscle couplon structure and microdomain Ca2+ signalling: a path towards ventricular arrhythmias","volume":"98","author":"Chopra","year":"2013","journal-title":"Cardiovasc. Res."},{"key":"10.1016\/j.stemcr.2023.04.005_bib7","doi-asserted-by":"crossref","first-page":"7636","DOI":"10.1073\/pnas.0902919106","article-title":"Ablation of triadin causes loss of cardiac Ca2+ release units, impaired excitation-contraction coupling, and cardiac arrhythmias","volume":"106","author":"Chopra","year":"2009","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"10.1016\/j.stemcr.2023.04.005_bib8","doi-asserted-by":"crossref","DOI":"10.1161\/CIRCGEN.119.002731","article-title":"Triadin knockout syndrome is absent in a multi-center molecular autopsy cohort of sudden infant death syndrome and sudden unexplained death in the young and is extremely rare in the general population","volume":"13","author":"Clemens","year":"2020","journal-title":"Circ. Genom. Precis. Med."},{"key":"10.1016\/j.stemcr.2023.04.005_bib9","doi-asserted-by":"crossref","DOI":"10.1161\/CIRCGEN.118.002419","article-title":"International Triadin Knockout Syndrome Registry: the clinical phenotype and treatment outcomes of patients with triadin knockout syndrome","volume":"12","author":"Clemens","year":"2019","journal-title":"Circ. Genom. Precis. Med."},{"key":"10.1016\/j.stemcr.2023.04.005_bib10","doi-asserted-by":"crossref","first-page":"1017","DOI":"10.1016\/j.hrthm.2020.01.012","article-title":"Phenotype-guided whole genome analysis in a patient with genetically elusive long QT syndrome yields a novel TRDN-encoded triadin pathogenetic substrate for triadin knockout syndrome and reveals a novel primate-specific cardiac TRDN transcript","volume":"17","author":"Clemens","year":"2020","journal-title":"Heart Rhythm"},{"key":"10.1016\/j.stemcr.2023.04.005_bib11","doi-asserted-by":"crossref","first-page":"1493","DOI":"10.1089\/scd.2009.0052","article-title":"Absence of transverse tubules contributes to non-uniform Ca2+ wavefronts in mouse and human embryonic stem cell\u2013derived cardiomyocytes","volume":"18","author":"Lieu","year":"2009","journal-title":"Stem Cells Dev."},{"key":"10.1016\/j.stemcr.2023.04.005_bib12","doi-asserted-by":"crossref","first-page":"1153","DOI":"10.1212\/WNL.0000000000003745","article-title":"Congenital myopathy associated with the triadin knockout syndrome","volume":"88","author":"Engel","year":"2017","journal-title":"Neurology"},{"key":"10.1016\/j.stemcr.2023.04.005_bib13","doi-asserted-by":"crossref","DOI":"10.1161\/CIRCGEN.119.002534","article-title":"Characterization of the CACNA1C-R518C missense mutation in the pathobiology of long-QT syndrome using human induced pluripotent stem cell cardiomyocytes shows action potential prolongation and L-type calcium channel perturbation","volume":"12","author":"Estes","year":"2019","journal-title":"Circ. Genom. Precis. Med."},{"key":"10.1016\/j.stemcr.2023.04.005_bib14","doi-asserted-by":"crossref","first-page":"9027","DOI":"10.1074\/jbc.270.16.9027","article-title":"Association of triadin with the ryanodine receptor and calsequestrin in the lumen of the sarcoplasmic reticulum","volume":"270","author":"Guo","year":"1995","journal-title":"J.\u00a0Biol. Chem."},{"key":"10.1016\/j.stemcr.2023.04.005_bib15","doi-asserted-by":"crossref","first-page":"458","DOI":"10.1074\/jbc.271.1.458","article-title":"Biochemical characterization and molecular cloning of cardiac triadin","volume":"271","author":"Guo","year":"1996","journal-title":"J.\u00a0Biol. Chem."},{"key":"10.1016\/j.stemcr.2023.04.005_bib16","doi-asserted-by":"crossref","first-page":"990","DOI":"10.1016\/j.jacc.2012.02.066","article-title":"Modeling of catecholaminergic polymorphic ventricular tachycardia with patient-specific human-induced pluripotent stem cells","volume":"60","author":"Itzhaki","year":"2012","journal-title":"J.\u00a0Am. Coll. Cardiol."},{"key":"10.1016\/j.stemcr.2023.04.005_bib17","doi-asserted-by":"crossref","first-page":"225","DOI":"10.1038\/nature09747","article-title":"Modelling the long QT syndrome with induced pluripotent stem cells","volume":"471","author":"Itzhaki","year":"2011","journal-title":"Nature"},{"key":"10.1016\/j.stemcr.2023.04.005_bib18","doi-asserted-by":"crossref","first-page":"3081","DOI":"10.1113\/jphysiol.2009.172098","article-title":"New roles of calsequestrin and triadin in cardiac muscle","volume":"587","author":"Knollmann","year":"2009","journal-title":"J.\u00a0Physiol."},{"key":"10.1016\/j.stemcr.2023.04.005_bib19","doi-asserted-by":"crossref","first-page":"17639","DOI":"10.1074\/jbc.M002091200","article-title":"Localization and characterization of the calsequestrin-binding domain of triadin 1. Evidence for a charged beta-strand in mediating the protein-protein interaction","volume":"275","author":"Kobayashi","year":"2000","journal-title":"J.\u00a0Biol. Chem."},{"key":"10.1016\/j.stemcr.2023.04.005_bib20","doi-asserted-by":"crossref","first-page":"1378","DOI":"10.1086\/324565","article-title":"A missense mutation in a highly conserved region of CASQ2 is associated with autosomal recessive catecholamine-induced polymorphic ventricular tachycardia in Bedouin families from Israel","volume":"69","author":"Lahat","year":"2001","journal-title":"Am. J. Hum. Genet."},{"key":"10.1016\/j.stemcr.2023.04.005_bib21","doi-asserted-by":"crossref","first-page":"2140","DOI":"10.1016\/j.jacc.2020.02.067","article-title":"KCNQ1 antibodies for immunotherapy of long QT syndrome type 2","volume":"75","author":"Maguy","year":"2020","journal-title":"J.\u00a0Am. Coll. Cardiol."},{"key":"10.1016\/j.stemcr.2023.04.005_bib22","doi-asserted-by":"crossref","first-page":"3261","DOI":"10.1113\/jphysiol.2014.281147","article-title":"Triadin regulation of the ryanodine receptor complex","volume":"593","author":"Marty","year":"2015","journal-title":"J.\u00a0Physiol."},{"key":"10.1016\/j.stemcr.2023.04.005_bib23","doi-asserted-by":"crossref","first-page":"514","DOI":"10.1016\/j.hrcr.2018.07.014","article-title":"A unique triadin exon deletion causing a null phenotype","volume":"4","author":"O\u2019Callaghan","year":"2018","journal-title":"HeartRhythm Case Rep."},{"key":"10.1016\/j.stemcr.2023.04.005_bib24","doi-asserted-by":"crossref","first-page":"34918","DOI":"10.1074\/jbc.M109.022442","article-title":"Triadin deletion induces impaired skeletal muscle function","volume":"284","author":"Oddoux","year":"2009","journal-title":"J.\u00a0Biol. Chem."},{"key":"10.1016\/j.stemcr.2023.04.005_bib25","doi-asserted-by":"crossref","first-page":"2281","DOI":"10.1038\/nprot.2013.143","article-title":"Genome engineering using the CRISPR-Cas9 system","volume":"8","author":"Ran","year":"2013","journal-title":"Nat. Protoc."},{"key":"10.1016\/j.stemcr.2023.04.005_bib26","doi-asserted-by":"crossref","first-page":"298","DOI":"10.1161\/CIRCRESAHA.108.171660","article-title":"Unexpected structural and functional consequences of the R33Q homozygous mutation in cardiac calsequestrin","volume":"103","author":"Rizzi","year":"2008","journal-title":"Circ. Res."},{"key":"10.1016\/j.stemcr.2023.04.005_bib27","doi-asserted-by":"crossref","first-page":"1146","DOI":"10.1111\/jce.12763","article-title":"New family with catecholaminergic polymorphic ventricular tachycardia linked to the triadin gene","volume":"26","author":"Rooryck","year":"2015","journal-title":"J.\u00a0Cardiovasc. Electrophysiol."},{"key":"10.1016\/j.stemcr.2023.04.005_bib28","doi-asserted-by":"crossref","first-page":"296","DOI":"10.1016\/j.hrthm.2019.08.018","article-title":"A novel homozygous mutation in the TRDN gene causes a severe form of pediatric malignant ventricular arrhythmia","volume":"17","author":"Rossi","year":"2019","journal-title":"Heart Rhythm"},{"key":"10.1016\/j.stemcr.2023.04.005_bib29","doi-asserted-by":"crossref","first-page":"2759","DOI":"10.1093\/hmg\/dds104","article-title":"Absence of triadin, a protein of the calcium release complex, is responsible for cardiac arrhythmia with sudden death in human","volume":"21","author":"Roux-Buisson","year":"2012","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.stemcr.2023.04.005_bib30","doi-asserted-by":"crossref","first-page":"58","DOI":"10.1038\/s41572-020-0188-7","article-title":"Inherited cardiac arrhythmias","volume":"6","author":"Schwartz","year":"2020","journal-title":"Nat. Rev. Dis. Primers"},{"key":"10.1016\/j.stemcr.2023.04.005_bib31","doi-asserted-by":"crossref","first-page":"34","DOI":"10.1016\/j.yjmcc.2014.12.025","article-title":"Transitions of protein traffic from cardiac ER to junctional SR","volume":"81","author":"Sleiman","year":"2015","journal-title":"J.\u00a0Mol. Cell. Cardiol."},{"key":"10.1016\/j.stemcr.2023.04.005_bib32","doi-asserted-by":"crossref","first-page":"497","DOI":"10.1111\/pace.12813","article-title":"Compound heterozygous triadin mutation causing cardiac arrest in two siblings","volume":"39","author":"Walsh","year":"2016","journal-title":"Pacing Clin. Electrophysiol."},{"key":"10.1016\/j.stemcr.2023.04.005_bib33","doi-asserted-by":"crossref","first-page":"320","DOI":"10.1016\/j.yjmcc.2017.12.002","article-title":"Hypertrophic cardiomyopathy-linked mutation in troponin T causes myofibrillar disarray and pro-arrhythmic action potential changes in human iPSC cardiomyocytes","volume":"114","author":"Wang","year":"2018","journal-title":"J.\u00a0Mol. Cell. Cardiol."}],"container-title":["Stem Cell Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S221367112300139X?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S221367112300139X?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,11,1]],"date-time":"2025-11-01T09:50:51Z","timestamp":1761990651000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S221367112300139X"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,5]]},"references-count":33,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2023,5]]}},"alternative-id":["S221367112300139X"],"URL":"https:\/\/doi.org\/10.1016\/j.stemcr.2023.04.005","relation":{},"ISSN":["2213-6711"],"issn-type":[{"value":"2213-6711","type":"print"}],"subject":[],"published":{"date-parts":[[2023,5]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Cellular and electrophysiological characterization of triadin knockout syndrome using induced pluripotent stem cell-derived cardiomyocytes","name":"articletitle","label":"Article Title"},{"value":"Stem Cell Reports","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.stemcr.2023.04.005","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2023 The Author(s).","name":"copyright","label":"Copyright"}]}}