{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,23]],"date-time":"2026-08-23T15:22:50Z","timestamp":1787498570318,"version":"build-2736575974"},"reference-count":24,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"5","license":[{"start":{"date-parts":[[2021,2,17]],"date-time":"2021-02-17T00:00:00Z","timestamp":1613520000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2021,2,17]],"date-time":"2021-02-17T00:00:00Z","timestamp":1613520000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":["Liver Transpl."],"published-print":{"date-parts":[[2021,5]]},"abstract":"<jats:p>Biliary atresia (BA) is the leading indication to perform a pediatric liver transplantation (LT). Timely hepatoportoenterostomy (HPE) attempts to interrupt the natural history and allow for enteric bile flow; however, most patients who are treated with HPE require LT by the age of 10 years. We determined the cost\u2010effectiveness of foregoing HPE to perform primary LT (pLT) in children with BA compared with standard\u2010of\u2010care HPE management. A Markov model was developed to simulate BA treatment over 10\u00a0years. Costs were measured in 2018 US dollars and effectiveness in life\u2010years (LYs). The primary outcome was incremental cost\u2010effectiveness ratio (ICER) between treatments. Model parameters were derived from the literature. In the base model, we assumed similar LT outcomes after HPE and pLT. Sensitivity analyses on all model parameters were performed, including a scenario in which pLT led to 100% patient and graft survival after LT. Children undergoing HPE accumulated $316,692 in costs and 8.17 LYs per patient. Children undergoing pLT accumulated $458,059 in costs and 8.24 LYs per patient, costing $1,869,164 per LY gained compared with HPE. With parameter variation over plausible ranges, only post\u2010HPE and post\u2010LT costs reduced the ICER below a typical threshold of $100,000 per LY gained. On probabilistic sensitivity analysis, 93% of iterations favored HPE at that threshold. With 100% patient and graft survival after pLT, pLT cost $283,478 per LY gained. HPE is more economically favorable than pLT for BA. pLT is unfavorable even with no graft or patient loss. The ability to predict those patients who may experience high costs after HPE or low costs after LT may help identify those patients for whom pLT could be considered.<\/jats:p>","DOI":"10.1002\/lt.25984","type":"journal-article","created":{"date-parts":[[2021,1,18]],"date-time":"2021-01-18T13:49:44Z","timestamp":1610977784000},"page":"711-718","update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":13,"title":["Cost\u2010Effectiveness of Primary Liver Transplantation Versus Hepatoportoenterostomy in the Management of Biliary Atresia in the United States"],"prefix":"10.1097","volume":"27","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6555-9732","authenticated-orcid":false,"given":"Vikram K.","family":"Raghu","sequence":"first","affiliation":[{"name":"Division of Gastroenterology, Hepatology, and Nutrition,University of Pittsburgh Medical Center Children\u2019s Hospital of Pittsburgh,Pittsburgh,PA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James E.","family":"Squires","sequence":"additional","affiliation":[{"name":"Division of Gastroenterology, Hepatology, and Nutrition,University of Pittsburgh Medical Center Children\u2019s Hospital of Pittsburgh,Pittsburgh,PA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Douglas B.","family":"Mogul","sequence":"additional","affiliation":[{"name":"Department of Pediatrics,Johns Hopkins University School of Medicine,Baltimore,MD"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Robert H.","family":"Squires","sequence":"additional","affiliation":[{"name":"Division of Gastroenterology, Hepatology, and Nutrition,University of Pittsburgh Medical Center Children\u2019s Hospital of Pittsburgh,Pittsburgh,PA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Patrick J.","family":"McKiernan","sequence":"additional","affiliation":[{"name":"Division of Gastroenterology, Hepatology, and Nutrition,University of Pittsburgh Medical Center Children\u2019s Hospital of Pittsburgh,Pittsburgh,PA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2624-8632","authenticated-orcid":false,"given":"George V.","family":"Mazariegos","sequence":"additional","affiliation":[{"name":"Thomas E. Starzl Transplantation Institute,Hillman Center for Pediatric Transplantation,Department of Transplant Surgery,University of Pittsburgh Medical Center Children\u2019s Hospital of Pittsburgh,Pittsburgh,PA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kenneth J.","family":"Smith","sequence":"additional","affiliation":[{"name":"Division of General Internal Medicine,University of Pittsburgh School of Medicine,Pittsburgh,PA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","published-online":{"date-parts":[[2021,2,17]]},"reference":[{"key":"lt25984-bib-0001-20250824","doi-asserted-by":"crossref","first-page":"396","DOI":"10.1097\/MPG.0000000000002450","article-title":"Biliary atresia as a disease starting in utero: implications for treatment, diagnosis, and pathogenesis","volume":"69","year":"2019","journal-title":"J Pediatr Gastroenterol Nutr"},{"key":"lt25984-bib-0002-20250824","doi-asserted-by":"crossref","first-page":"1127","DOI":"10.1016\/j.jpedsurg.2019.02.051","article-title":"Predictors of need for liver transplantation in children undergoing hepatoportoenterostomy for biliary atresia","volume":"54","year":"2019","journal-title":"J Pediatr Surg"},{"issue":"suppl 1","key":"lt25984-bib-0003-20250824","first-page":"193","article-title":"OPTN\/SRTR 2018 annual data report: liver","volume":"20","year":"2020","journal-title":"Am J Transplant"},{"key":"lt25984-bib-0004-20250824","doi-asserted-by":"crossref","first-page":"1297","DOI":"10.1007\/s00383-017-4174-4","article-title":"Biliary atresia and liver transplantation: results and thoughts for primary liver transplantation in select patients","volume":"33","year":"2017","journal-title":"Pediatr Surg Int"},{"key":"lt25984-bib-0005-20250824","doi-asserted-by":"crossref","first-page":"751","DOI":"10.1002\/hep.30935","article-title":"Biliary atresia: biliary\u2010enteric drainage or primary liver transplant?","volume":"71","year":"2020","journal-title":"Hepatology"},{"key":"lt25984-bib-0006-20250824","doi-asserted-by":"crossref","first-page":"26","DOI":"10.1001\/jamasurg.2018.3180","article-title":"Biliary\u2010enteric drainage vs primary liver transplant as initial treatment for children with biliary atresia","volume":"154","year":"2019","journal-title":"JAMA Surg"},{"key":"lt25984-bib-0007-20250824","article-title":"Expanding analytic possibilities in pediatric solid organ transplantation through linkage of administrative and clinical registry databases","volume":"23","year":"2019","journal-title":"Pediatr Transplant"},{"key":"lt25984-bib-0008-20250824","doi-asserted-by":"crossref","first-page":"237","DOI":"10.1016\/j.jpeds.2018.04.001","article-title":"Factors associated with timing and adverse outcomes in patients with biliary atresia undergoing Kasai hepatoportoenterostomy","volume":"199","year":"2018","journal-title":"J Pediatr"},{"key":"lt25984-bib-0009-20250824","doi-asserted-by":"crossref","first-page":"678","DOI":"10.1177\/0272989X12454941","article-title":"Conceptualizing a model: a report of the ISPOR\u2010SMDM Modeling Good Research Practices Task Force\u20102","volume":"32","year":"2012","journal-title":"Med Decis Making"},{"key":"lt25984-bib-0010-20250824","doi-asserted-by":"crossref","first-page":"91","DOI":"10.1097\/MPG.0000000000000569","article-title":"Cost\u2010effective analysis of screening for biliary atresia with the stool color card","volume":"60","year":"2015","journal-title":"J Pediatr Gastroenterol Nutr"},{"key":"lt25984-bib-0011-20250824","doi-asserted-by":"crossref","first-page":"89","DOI":"10.1016\/j.jpeds.2019.12.023","article-title":"Improved outcomes for liver transplantation in patients with biliary atresia since pediatric end\u2010stage liver disease implementation: analysis of the Society of Pediatric Liver Transplantation Registry","volume":"219","year":"2020","journal-title":"J Pediatr"},{"key":"lt25984-bib-0012-20250824","doi-asserted-by":"crossref","first-page":"220","DOI":"10.1097\/MPG.0000000000000986","article-title":"Liver transplantation for biliary atresia: is there a difference in outcome for infants?","volume":"62","year":"2016","journal-title":"J Pediatr Gastroenterol Nutr"},{"key":"lt25984-bib-0014-20250824","doi-asserted-by":"crossref","first-page":"796","DOI":"10.1056\/NEJMp1405158","article-title":"Updating cost\u2010effectiveness\u2014the curious resilience of the $50,000\u2010per\u2010QALY threshold","volume":"371","year":"2014","journal-title":"N Engl J Med"},{"key":"lt25984-bib-0016-20250824","first-page":"4","article-title":"Pathogenesis and outcome of biliary atresia: current concepts","volume":"37","year":"2003","journal-title":"J Pediatr Gastroenterol Nutr"},{"key":"lt25984-bib-0017-20250824","first-page":"1","article-title":"Births: final data for 2018","volume":"68","year":"2019","journal-title":"Natl Vital Stat Rep"},{"key":"lt25984-bib-0018-20250824","doi-asserted-by":"crossref","first-page":"584","DOI":"10.1177\/0272989X15578125","article-title":"Computing expected value of partial sample information from probabilistic sensitivity analysis using linear regression metamodeling","volume":"35","year":"2015","journal-title":"Med Decis Making"},{"key":"lt25984-bib-0019-20250824","doi-asserted-by":"crossref","first-page":"1280","DOI":"10.1542\/peds.2008-1949","article-title":"Impact of age at Kasai operation on its results in late childhood and adolescence: a rational basis for biliary atresia screening","volume":"123","year":"2009","journal-title":"Pediatrics"},{"key":"lt25984-bib-0020-20250824","article-title":"Determinants of length of stay after pediatric liver transplantation","volume":"24","year":"2020","journal-title":"Pediatr Transplant"},{"key":"lt25984-bib-0021-20250824","doi-asserted-by":"crossref","first-page":"86","DOI":"10.1002\/lt.24646","article-title":"Global lessons in graft type and pediatric liver allocation: a path toward improving outcomes and eliminating wait\u2010list mortality","volume":"23","year":"2017","journal-title":"Liver Transpl"},{"key":"lt25984-bib-0022-20250824","doi-asserted-by":"crossref","first-page":"988","DOI":"10.1053\/j.gastro.2017.06.053","article-title":"Analysis of liver offers to pediatric candidates on the transplant wait list","volume":"153","year":"2017","journal-title":"Gastroenterology"},{"key":"lt25984-bib-0023-20250824","doi-asserted-by":"crossref","first-page":"472","DOI":"10.1097\/MPG.0000000000002287","article-title":"The impact of increased allocation priority for children awaiting liver transplant: a liver simulated allocation model (LSAM) analysis","volume":"68","year":"2019","journal-title":"J Pediatr Gastroenterol Nutr"},{"key":"lt25984-bib-0024-20250824","doi-asserted-by":"crossref","first-page":"1627","DOI":"10.1097\/TP.0000000000003079","article-title":"Impact of acuity circles on outcomes for pediatric liver transplant candidates","volume":"104","year":"2020","journal-title":"Transplantation"},{"key":"lt25984-bib-0025-20250824","doi-asserted-by":"crossref","first-page":"203","DOI":"10.1097\/MPG.0000000000001289","article-title":"Early posthepatoportoenterostomy predictors of native liver survival in biliary atresia","volume":"64","year":"2017","journal-title":"J Pediatr Gastroenterol Nutr"},{"key":"lt25984-bib-0026-20250824","article-title":"Health utility and quality of life in pediatric liver transplant recipients","volume":"24","year":"2020","journal-title":"Pediatr Transplant"}],"container-title":["Liver Transplantation"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/lt.25984","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/lt.25984","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.lww.com\/10.1002\/lt.25984","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,24]],"date-time":"2025-08-24T21:04:15Z","timestamp":1756069455000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.lww.com\/10.1002\/lt.25984"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,2,17]]},"references-count":24,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2021]]}},"URL":"https:\/\/doi.org\/10.1002\/lt.25984","relation":{},"ISSN":["1527-6465","1527-6473"],"issn-type":[{"value":"1527-6465","type":"print"},{"value":"1527-6473","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,2,17]]},"assertion":[{"value":"2020-11-17","name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2021-01-04","name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}}]}}