{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T20:44:11Z","timestamp":1790801051274,"version":"4.1.0"},"reference-count":13,"publisher":"Elsevier BV","issue":"10","license":[{"start":{"date-parts":[[2022,10,1]],"date-time":"2022-10-01T00:00:00Z","timestamp":1664582400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2022,10,1]],"date-time":"2022-10-01T00:00:00Z","timestamp":1664582400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2023,9,29]],"date-time":"2023-09-29T00:00:00Z","timestamp":1695945600000},"content-version":"am","delay-in-days":363,"URL":"http:\/\/www.elsevier.com\/open-access\/userlicense\/1.0\/"}],"funder":[{"DOI":"10.13039\/100000050","name":"National Heart, Lung, and Blood Institute","doi-asserted-by":"publisher","award":["K23HL130554"],"award-info":[{"award-number":["K23HL130554"]}],"id":[{"id":"10.13039\/100000050","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000050","name":"National Heart, Lung, and Blood Institute","doi-asserted-by":"publisher","award":["K23HL136932"],"award-info":[{"award-number":["K23HL136932"]}],"id":[{"id":"10.13039\/100000050","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000050","name":"National Heart, Lung, and Blood Institute","doi-asserted-by":"publisher","award":["R01HL136389"],"award-info":[{"award-number":["R01HL136389"]}],"id":[{"id":"10.13039\/100000050","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000050","name":"National Heart, Lung, and Blood Institute","doi-asserted-by":"publisher","award":["R01HL147108"],"award-info":[{"award-number":["R01HL147108"]}],"id":[{"id":"10.13039\/100000050","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000968","name":"American Heart Association","doi-asserted-by":"publisher","award":["17SDG33410183"],"award-info":[{"award-number":["17SDG33410183"]}],"id":[{"id":"10.13039\/100000968","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["clinicalkey.com","clinicalkey.com.au","clinicalkey.es","clinicalkey.fr","clinicalkey.jp","heartrhythmjournal.com","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Heart Rhythm"],"published-print":{"date-parts":[[2022,10]]},"DOI":"10.1016\/j.hrthm.2022.05.009","type":"journal-article","created":{"date-parts":[[2022,5,11]],"date-time":"2022-05-11T13:33:07Z","timestamp":1652275987000},"page":"1673-1681","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":36,"title":["Cardiac crises: Cardiac arrhythmias and cardiomyopathy during TANGO2 deficiency related metabolic crises"],"prefix":"10.1016","volume":"19","author":[{"given":"Christina Y.","family":"Miyake","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Erica J.","family":"Lay","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cheyenne M.","family":"Beach","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Scott R.","family":"Ceresnak","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Caridad M.","family":"Delauz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Taylor S.","family":"Howard","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christopher M.","family":"Janson","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kate","family":"Jardine","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Prince J.","family":"Kannankeril","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maina","family":"Kava","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeffrey J.","family":"Kim","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leonardo","family":"Liberman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Scott L.","family":"Macicek","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tam Dam","family":"Pham","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Terry","family":"Robertson","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Santiago O.","family":"Valdes","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gregory","family":"Webster","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sara B.","family":"Stephens","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Diana M.","family":"Milewicz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mahshid","family":"Azamian","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Saad A.","family":"Ehsan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kimberly M.","family":"Houck","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Claudia","family":"Soler-Alfonso","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kevin E.","family":"Glinton","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mustafa","family":"Tosur","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Na","family":"Li","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Weiyi","family":"Xu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Seema R.","family":"Lalani","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lilei","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.hrthm.2022.05.009_bib1","doi-asserted-by":"crossref","first-page":"347","DOI":"10.1016\/j.ajhg.2015.12.008","article-title":"Recurrent muscle weakness with rhabdomyolysis, metabolic crises, and cardiac arrhythmia due to bi-allelic TANGO2 mutations","volume":"98","author":"Lalani","year":"2016","journal-title":"Am J Hum Genet"},{"key":"10.1016\/j.hrthm.2022.05.009_bib2","doi-asserted-by":"crossref","first-page":"358","DOI":"10.1016\/j.ajhg.2015.12.009","article-title":"Bi-allelic truncating mutations in TANGO2 cause infancy-onset recurrent metabolic crises with encephalocardiomyopathy","volume":"98","author":"Kremer","year":"2016","journal-title":"Am J Hum Genet"},{"key":"10.1016\/j.hrthm.2022.05.009_bib3","doi-asserted-by":"crossref","first-page":"415","DOI":"10.1002\/jimd.12314","article-title":"Clinical and biological characterization of 20 patients with TANGO2 deficiency indicates novel triggers of metabolic crises and no primary energetic defect","volume":"44","author":"Berat","year":"2021","journal-title":"J Inherit Metab Dis"},{"key":"10.1016\/j.hrthm.2022.05.009_bib4","doi-asserted-by":"crossref","first-page":"601","DOI":"10.1038\/s41436-018-0137-y","article-title":"TANGO2: expanding the clinical phenotype and spectrum of pathogenic variants","volume":"21","author":"Dines","year":"2019","journal-title":"Genet Med"},{"key":"10.1016\/j.hrthm.2022.05.009_bib5","doi-asserted-by":"crossref","first-page":"898","DOI":"10.1002\/jimd.12149","article-title":"TANGO2 deficiency as a cause of neurodevelopmental delay with indirect effects on mitochondrial energy metabolism","volume":"42","author":"Jennions","year":"2019","journal-title":"J Inherit Metab Dis"},{"key":"10.1016\/j.hrthm.2022.05.009_bib6","doi-asserted-by":"crossref","first-page":"473","DOI":"10.1002\/ajmg.a.62543","article-title":"Variable clinical severity in TANGO2 deficiency: case series and literature review","volume":"188","author":"Schymick","year":"2022","journal-title":"Am J Med Genet A"},{"key":"10.1016\/j.hrthm.2022.05.009_bib7","doi-asserted-by":"crossref","first-page":"80","DOI":"10.1016\/j.arcped.2020.11.004","article-title":"Clinical phenotype associated with TANGO2 gene mutation","volume":"28","author":"Hoebeke","year":"2021","journal-title":"Arch Pediatr"},{"key":"10.1016\/j.hrthm.2022.05.009_bib8","doi-asserted-by":"crossref","first-page":"297","DOI":"10.1002\/jimd.12156","article-title":"Clinical presentation and proteomic signature of patients with TANGO2 mutations","volume":"43","author":"Mingirulli","year":"2020","journal-title":"J Inherit Metab Dis"},{"key":"10.1016\/j.hrthm.2022.05.009_bib9","doi-asserted-by":"crossref","first-page":"34","DOI":"10.1016\/j.pediatrneurol.2021.02.011","article-title":"Symptom prevalence and genotype-phenotype correlations in patients with TANGO2-related metabolic encephalopathy and arrhythmias (TRMEA)","volume":"119","author":"Powell","year":"2021","journal-title":"Pediatr Neurol"},{"key":"10.1016\/j.hrthm.2022.05.009_bib10","unstructured":"Lalani S, Graham B, Burrage L, et\u00a0al. TANGO2-related metabolic encephalopathy and arrhythmias. 2018 Jan 25 [updated 2018 Dec 20]. In: Adam MP, Ardinger HH, Pagon RA, et\u00a0al, Editors. GeneReviews [Internet]. Seattle, WA: University of Washington, Seattle; 1993\u20132022."},{"key":"10.1016\/j.hrthm.2022.05.009_bib11","doi-asserted-by":"crossref","first-page":"256","DOI":"10.1016\/j.hrcr.2020.01.007","article-title":"Electrical storm treated successfully in a patient with TANGO2 gene mutation and long QT syndrome: a case report","volume":"6","author":"Scuotto","year":"2020","journal-title":"HeartRhythm Case Rep"},{"key":"10.1016\/j.hrthm.2022.05.009_bib12","doi-asserted-by":"crossref","DOI":"10.1007\/BF03086296","article-title":"Founder mutations in the Netherlands: SCN5a 1795insD, the first described arrhythmia overlap syndrome and one of the largest and best characterised families worldwide","volume":"17","author":"Postema","year":"2009","journal-title":"Neth Heart J"},{"key":"10.1016\/j.hrthm.2022.05.009_bib13","doi-asserted-by":"crossref","first-page":"2584","DOI":"10.1161\/CIRCULATIONAHA.106.653949","article-title":"Overlap syndrome of cardiac sodium channel disease in mice carrying the equivalent mutation of human SCN5A-1795insD","volume":"114","author":"Remme","year":"2006","journal-title":"Circulation"}],"container-title":["Heart Rhythm"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S154752712201966X?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S154752712201966X?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,10,27]],"date-time":"2025-10-27T06:57:15Z","timestamp":1761548235000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S154752712201966X"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,10]]},"references-count":13,"journal-issue":{"issue":"10","published-print":{"date-parts":[[2022,10]]}},"alternative-id":["S154752712201966X"],"URL":"https:\/\/doi.org\/10.1016\/j.hrthm.2022.05.009","relation":{},"ISSN":["1547-5271"],"issn-type":[{"value":"1547-5271","type":"print"}],"subject":[],"published":{"date-parts":[[2022,10]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Cardiac crises: Cardiac arrhythmias and cardiomyopathy during TANGO2 deficiency related metabolic crises","name":"articletitle","label":"Article Title"},{"value":"Heart Rhythm","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.hrthm.2022.05.009","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2022 Published by Elsevier Inc. on behalf of Heart Rhythm Society.","name":"copyright","label":"Copyright"}]}}