{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,8]],"date-time":"2026-10-08T12:28:13Z","timestamp":1791462493270,"version":"4.3.4"},"reference-count":7,"publisher":"Springer Science and Business Media LLC","issue":"3","license":[{"start":{"date-parts":[[2022,12,2]],"date-time":"2022-12-02T00:00:00Z","timestamp":1669939200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2022,12,2]],"date-time":"2022-12-02T00:00:00Z","timestamp":1669939200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Pediatr Cardiol"],"published-print":{"date-parts":[[2023,3]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Neonatal lupus (NLE) is a rare acquired autoimmune disorder caused by transplacental passage of maternal autoantibodies to Sjogren\u2019s Syndrome A or B (SSA-SSB) autoantigens (Vanoni et al. in Clin Rev Allerg Immunol 53:469\u2013476, 2017) which target fetal and neonatal tissues for immune destruction. The cardiac trademark of NLE is autoimmune heart block, which accounts for more than 80% of cases of complete atrioventricular heart block (AVB) in newborns with a structurally normal heart (Martin in Cardiol Young 24: 41\u201346, 2014). NLE presenting with cardiac alterations not involving rhythm disturbances are described in the literature, but they are rare. Here, we report a case of a neonate with high anti-SSA antibodies who developed severe ventricular dysfunction in the absence of rhythm abnormalities, endocardial fibroelastosis, and dilated cardiomyopathy (Trucco et al. in J Am Coll Cardiol 57:715\u2013723, <jats:ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" ext-link-type=\"doi\" xlink:href=\"10.1016\/j.jacc.2010.09.044\">https:\/\/doi.org\/10.1016\/j.jacc.2010.09.044<\/jats:ext-link>, 2011), the most common cardiac presentations of NLE. The patient developed severe multiorgan dysfunction syndrome that required prolonged critical care support but fully recovered and was discharged home. We highlight the unusual clinical features of this NLE case and the importance of timely treatment of NLE allowing complete recovery of a critically ill neonate.\n<\/jats:p>","DOI":"10.1007\/s00246-022-03056-y","type":"journal-article","created":{"date-parts":[[2022,12,2]],"date-time":"2022-12-02T17:03:48Z","timestamp":1670000628000},"page":"736-739","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["A Case of Neonatal Lupus Presenting with Myocardial Dysfunction in the Absence of Congenital Heart Block (CHB): Clinical Management and Brief Literature Review of Neonatal Cardiac Lupus"],"prefix":"10.1007","volume":"44","author":[{"given":"Samhita","family":"Jain","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ruggero","family":"Spadafora","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarah","family":"Maxwell","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carlos","family":"Botas","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hythem","family":"Nawaytou","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emily","family":"von Scheven","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elizabeth E.","family":"Crouch","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2022,12,2]]},"reference":[{"key":"3056_CR1","doi-asserted-by":"publisher","first-page":"469","DOI":"10.1007\/s12016-017-8653-0","volume":"53","author":"F Vanoni","year":"2017","unstructured":"Vanoni F, Lava SAG, Fossali EF, Cavalli R, Simonetti GD, Bianchetti MG, Bozzini M-A, Agostoni C, Milani GP (2017) Neonatal systemic lupus erythematous syndrome: a comprehensive review. Clin Rev Allerg Immunol 53:469\u2013476","journal-title":"Clin Rev Allerg Immunol"},{"issue":"supp.2","key":"3056_CR2","doi-asserted-by":"publisher","first-page":"41","DOI":"10.1017\/S1047951114001358","volume":"24","author":"TA Martin","year":"2014","unstructured":"Martin TA (2014) Congenital heart block: current thoughts on management, morphologic spectrum, and role of intervention. Cardiol Young 24(supp.2):41\u201346","journal-title":"Cardiol Young"},{"issue":"6","key":"3056_CR3","doi-asserted-by":"publisher","first-page":"715","DOI":"10.1016\/j.jacc.2010.09.044","volume":"57","author":"SM Trucco","year":"2011","unstructured":"Trucco SM, Jaeggi E, Cuneo B, Moon-Grady AJ, Silverman E, Silverman N, Hornberger LK (2011) Use of intravenous gamma globulin and corticosteroids in the treatment of maternal autoantibody-mediated cardiomyopathy. J Am Coll Cardiol 57(6):715\u2013723. https:\/\/doi.org\/10.1016\/j.jacc.2010.09.044","journal-title":"J Am Coll Cardiol"},{"key":"3056_CR4","doi-asserted-by":"publisher","first-page":"N4","DOI":"10.1016\/S0735-1097(02)02004-1","volume":"40","author":"LE Nield","year":"2002","unstructured":"Nield LE, Silverman E, Smallhorn JF, Taylor GP, Brendan J, Mullen M, Benson LN, Hornberger LK (2002) Endocardial fibroelastosis associated with maternal anti-Ro and anti-La antibodies in the absence of atrioventricular block. J Am Coll Cardiol 40:N4","journal-title":"J Am Coll Cardiol"},{"key":"3056_CR5","doi-asserted-by":"publisher","first-page":"189","DOI":"10.1111\/j.1365-3083.2010.02437.x","volume":"72","author":"LK Hornberger","year":"2010","unstructured":"Hornberger LK, Al Rajaa N (2010) Spectrum of cardiac involvement in neonatal lupus. Scand J Immunol 72:189\u2013197","journal-title":"Scand J Immunol"},{"key":"3056_CR6","doi-asserted-by":"publisher","first-page":"N1","DOI":"10.1542\/peds.106.1.86","volume":"106","author":"M Eronen","year":"2000","unstructured":"Eronen M, Siren M-K, Ekblad H, Tikanoja T, Julkunen H, Paavilainen T (2000) Short and long-term outcome of children with congenital complete heart block diagnosed in utero or as newborn. Pediatrics 106:N1","journal-title":"Pediatrics"},{"key":"3056_CR7","doi-asserted-by":"publisher","first-page":"1154","DOI":"10.1016\/j.autrev.2015.08.005","volume":"14","author":"K Levesque","year":"2015","unstructured":"Levesque K, Morel N, Maltret A, Baron G, Masseau A, Otquevaux P, Piette J-C, Barrere F, Le Bidois J, Fermont L, Fain O et al (2015) Description of 214 cases of autoimmune congenital heart block: results of the French neonatal lupus syndrome. Autoimmun Rev 14:1154\u20131216","journal-title":"Autoimmun Rev"}],"container-title":["Pediatric Cardiology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s00246-022-03056-y.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s00246-022-03056-y\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s00246-022-03056-y.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,2,23]],"date-time":"2023-02-23T09:07:21Z","timestamp":1677143241000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s00246-022-03056-y"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,12,2]]},"references-count":7,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2023,3]]}},"alternative-id":["3056"],"URL":"https:\/\/doi.org\/10.1007\/s00246-022-03056-y","relation":{},"ISSN":["0172-0643","1432-1971"],"issn-type":[{"value":"0172-0643","type":"print"},{"value":"1432-1971","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,12,2]]},"assertion":[{"value":"17 August 2022","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"16 November 2022","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"2 December 2022","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Authors have no conflict of interest to declare.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}},{"value":"Informed consent was obtained from all individual participants included in the study.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Informed Consent"}}]}}