{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2024,5,20]],"date-time":"2024-05-20T04:57:55Z","timestamp":1716181075372},"reference-count":14,"publisher":"Georg Thieme Verlag KG","issue":"04","license":[{"start":{"date-parts":[[2020,12,3]],"date-time":"2020-12-03T00:00:00Z","timestamp":1606953600000},"content-version":"vor","delay-in-days":63,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["AJP Rep"],"published-print":{"date-parts":[[2020,10]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>\n          Introduction\u2003Congenital complete atrioventricular block (CCAVB) associated with congenital chylothorax is a rare finding that has been reported in only one case in the literature. We report here the case of an infant with CCAVB complicated by congenital chylothorax.<\/jats:p><jats:p>\n          Patient Report\u2003We present the case of a male neonate with a birth weight of 2114\u2009g. Fetal bradycardia and right pleural effusion were detected at gestational age of 22 weeks. Maternal serum levels of anti-Sj\u00f6gren's-syndrome-related antigen A autoantibody were high (4840 U\/mL). The neonate was delivered at gestational age of 33 weeks; a temporary external pacemaker was placed immediately after birth that resulted in an improved cardiac output. Milk-colored pleural effusion increased in volume together with the initiation of breast milk feeding. Lymphocytosis and high triglyceride levels in the pleural fluid led to the diagnosis of chylothorax. The pleural effusion resolved in response to prednisolone, octreotide, and total parenteral nutrition.<\/jats:p><jats:p>\n          Discussion\u2003The causal relationship between CCAVB and congenital chylothorax can be explained by considering the damage to the lymphatic vessels secondary to inflammation due to maternal autoantibodies and venous congestion due to bradycardia.<\/jats:p><jats:p>\n          Conclusion\u2003In any case of CCAVB associated with atypical pleural effusion, one must consider the possibility of congenital chylothorax.<\/jats:p>","DOI":"10.1055\/s-0040-1715178","type":"journal-article","created":{"date-parts":[[2020,12,4]],"date-time":"2020-12-04T00:03:38Z","timestamp":1607040218000},"page":"e403-e407","source":"Crossref","is-referenced-by-count":1,"title":["Chylothorax Associated with Congenital Complete Atrioventricular Block"],"prefix":"10.1055","volume":"10","author":[{"given":"Shuhei","family":"Fujino","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hidehiko","family":"Maruyama","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Keiko","family":"Tsukamoto","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hiroshi","family":"Ono","sequence":"additional","affiliation":[{"name":"Division of Cardiology, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tetsuya","family":"Isayama","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yushi","family":"Ito","sequence":"additional","affiliation":[{"name":"Division of Neonatology, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"194","published-online":{"date-parts":[[2020,12,3]]},"reference":[{"issue":"03","key":"ref1","first-page":"85","article-title":"Congenital complete heart block: an international study of the natural history","volume":"4","author":"M Micha\u00eblsson","year":"1972","journal-title":"Cardiovasc Clin"},{"key":"ref2","first-page":"695","volume-title":"Surgical Disorders of the Chest and Airways","author":"B A Keller","year":"2018"},{"key":"ref3","first-page":"1073","article-title":"A case of Down syndrome baby, with fetal hydrops and congenital complete AV block","volume":"49","author":"K Saito","year":"2013","journal-title":"J Jpn Soc Perin Neon Med"},{"issue":"06","key":"ref4","doi-asserted-by":"crossref","first-page":"585","DOI":"10.3109\/s10165-008-0100-x","article-title":"Anti-SS-A\/Ro antibody determination by indirect immunofluorescence and comparison of different methods of anti-nuclear antibody screening: 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Lin","year":"2007","journal-title":"Clin Rheumatol"},{"issue":"06","key":"ref12","first-page":"1330","article-title":"Concurrent occurrence of chylothorax, chylous ascites, and protein-losing enteropathy in systemic lupus erythematosus","volume":"29","author":"C K Lee","year":"2002","journal-title":"J Rheumatol"},{"key":"ref13","first-page":"279","article-title":"Case report of congenital chylothorax effectively treated by prednisolone administration","volume":"21","author":"T Dohmoto","year":"2009","journal-title":"Journal of Japan Society for Premature and Newborn Medicine"},{"issue":"01","key":"ref14","doi-asserted-by":"crossref","first-page":"56","DOI":"10.1007\/s00246-005-0993-5","article-title":"Plasmatic [corrected] factor XIII reduces severe pleural effusion in children after open-heart surgery","volume":"27","author":"M Schroth","year":"2006","journal-title":"Pediatr Cardiol"}],"container-title":["American Journal of Perinatology 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