{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,17]],"date-time":"2026-03-17T22:44:35Z","timestamp":1773787475296,"version":"3.50.1"},"reference-count":13,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2025,10,7]],"date-time":"2025-10-07T00:00:00Z","timestamp":1759795200000},"content-version":"vor","delay-in-days":279,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2025,1,1]],"date-time":"2025-01-01T00:00:00Z","timestamp":1735689600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Case Reports in Pediatrics"],"published-print":{"date-parts":[[2025,1]]},"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Inspissated bile syndrome (IBS) is a rare neonatal condition characterized by thickened bile leading to cholestasis, while Crigler\u2013Najjar syndrome type II (CNS\u2010II) is an autosomal recessive disorder resulting in unconjugated hyperbilirubinemia. Both conditions pose significant risks, particularly when they converge during pregnancy.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case Presentation<\/jats:title>\n                    <jats:p>We report a unique case involving a 20\u2009day\u2010old female neonate born to a mother with CNS\u2010II who discontinued phenobarbital treatment during pregnancy. The mother presented with elevated indirect bilirubin levels (total 32, and direct 0.4\u2009mg\/dL) and normal liver function test, while the neonate exhibited prolonged jaundice and cholestasis with direct hyperbilirubinemia (total 8, and direct 5.4\u2009mg\/dL) upon admission. Treatment included ursodeoxycholic acid for the neonate, leading to normalization of bilirubin levels. The neonate\u2019s growth and neurological development remained normal at 6\u2009months.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Discussion<\/jats:title>\n                    <jats:p>This case emphasizes the significant risks associated with untreated CNS\u2010II during pregnancy, which likely contributed to the neonate\u2019s IBS. Existing literature highlights the importance of maternal management in preventing severe neonatal complications associated with hyperbilirubinemia.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>This case highlights the importance of timely intervention in managing maternal metabolic disorders during pregnancy to improve neonatal health. Further research is essential to develop optimal treatment strategies for such cases.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1155\/crpe\/4964181","type":"journal-article","created":{"date-parts":[[2025,10,7]],"date-time":"2025-10-07T07:09:33Z","timestamp":1759820973000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Inspissated Bile Syndrome and Crigler\u2013Najjar Syndrome Type II: When Two Rare Conditions Converge"],"prefix":"10.1155","volume":"2025","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4496-1108","authenticated-orcid":false,"given":"Razieh","family":"Sangsari","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7659-3533","authenticated-orcid":false,"given":"Maryam","family":"Saeedi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2974-6362","authenticated-orcid":false,"given":"Kayvan","family":"Mirnia","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0003-5552-838X","authenticated-orcid":false,"given":"Fatemeh","family":"Tafreshi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2025,10,7]]},"reference":[{"key":"e_1_2_10_1_2","doi-asserted-by":"publisher","DOI":"10.4254\/wjh.v8.i11.530"},{"key":"e_1_2_10_2_2","volume-title":"Crigler-Najjar Syndrome","author":"Bhandari J.","year":"2024"},{"key":"e_1_2_10_3_2","doi-asserted-by":"publisher","DOI":"10.7860\/JCDR\/2017\/28195.10221"},{"key":"e_1_2_10_4_2","doi-asserted-by":"publisher","DOI":"10.1177\/1753495x221099443"},{"key":"e_1_2_10_5_2","doi-asserted-by":"publisher","DOI":"10.1159\/000238122"},{"key":"e_1_2_10_6_2","first-page":"168","article-title":"Crigler-Najjar Syndrome Type 2\u2013An Atypical Case","volume":"24","author":"Raposo F.","year":"2014","journal-title":"Clinical Case Reports"},{"key":"e_1_2_10_7_2","doi-asserted-by":"publisher","DOI":"10.3389\/fmed.2024.1354514"},{"key":"e_1_2_10_8_2","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2006-3669"},{"key":"e_1_2_10_9_2","doi-asserted-by":"publisher","DOI":"10.7759\/cureus.59075"},{"key":"e_1_2_10_10_2","volume-title":"Inspissated Bile Syndrome Mimicking Biliary Atresia:Effectiveness of the Laparoscopic Cholangiography With Therapeutic Saline Lavage Approach","author":"Atitallah S.","year":"2025"},{"key":"e_1_2_10_11_2","doi-asserted-by":"publisher","DOI":"10.3390\/ijms252011006"},{"key":"e_1_2_10_12_2","first-page":"1124","article-title":"Successful Photo-and Phenobarbital Therapy During Pregnancy in a Woman With Crigler-Najjar Syndrome Type II","volume":"40","author":"Holstein A.","year":"2005","journal-title":"Scandinavian Journal of Gastroenterology"},{"key":"e_1_2_10_13_2","doi-asserted-by":"crossref","DOI":"10.2399\/prn.23.0311007","article-title":"Management of a Pregnancy With Crigler-Najjar syndrome Type 2: A Case Report","author":"\u00c7a\u011fl\u0131yan E.","year":"2023","journal-title":"Perinatal Journal"}],"container-title":["Case Reports in Pediatrics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1155\/crpe\/4964181","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1155\/crpe\/4964181","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1155\/crpe\/4964181","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,3,17]],"date-time":"2026-03-17T19:06:30Z","timestamp":1773774390000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1155\/crpe\/4964181"}},"subtitle":[],"editor":[{"given":"Denis 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