{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,11,18]],"date-time":"2025-11-18T05:58:39Z","timestamp":1763445519656,"version":"3.37.3"},"reference-count":35,"publisher":"Springer Science and Business Media LLC","issue":"4","license":[{"start":{"date-parts":[[2022,2,15]],"date-time":"2022-02-15T00:00:00Z","timestamp":1644883200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2022,2,15]],"date-time":"2022-02-15T00:00:00Z","timestamp":1644883200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100004398","name":"Mitsubishi Foundation","doi-asserted-by":"publisher","award":["ID# 201930034"],"award-info":[{"award-number":["ID# 201930034"]}],"id":[{"id":"10.13039\/501100004398","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Pediatr Surg Int"],"published-print":{"date-parts":[[2022,4]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Background<\/jats:title>\n                <jats:p>Treatment of biliary atresia (BA), which typically requires an initial surgical intervention called the Kasai procedure (KP) and possible liver transplant (LT) afterwards, is quite resource-intensive and would affect patients and families for a lifetime; yet a comprehensive view of the economic burden has not been reported. We estimated direct health care costs from the public payer perspective using the National Database of Health Insurance Claims and Specific Health Checkups of Japan.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Methods<\/jats:title>\n                <jats:p>Children newly diagnosed at ages 0\u00a0days to 4\u00a0years between April 2010 and September 2019 were identified. Costs of treatment were estimated for six phases of care: prediagnosis, KP and inpatient hospitalization, follow-up after KP, pre-transplant checkup, LT and inpatient hospitalization, and follow-up after LT.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Results<\/jats:title>\n                <jats:p>Mean total prediagnosis medical cost was $6847 (USD) and KP and inpatient hospitalization was $42,157 per year. Follow-up after KP was $15,499, and pre-transplant checkup after KP was $36,015 per year. Mean cost for LT and inpatient hospitalization was $105,334, and follow-up after liver transplant was $25,459 per year.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusions<\/jats:title>\n                <jats:p>Treatment of BA requires extensive medical resource consumption. The use of the comprehensive national database allowed us to estimate the costs which will be useful for health service planning and cost-effectiveness analysis.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1007\/s00383-022-05079-1","type":"journal-article","created":{"date-parts":[[2022,2,15]],"date-time":"2022-02-15T04:39:42Z","timestamp":1644899982000},"page":"547-554","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Direct health care cost of treatment and medication of biliary atresia patients using the National Database of Health Insurance Claims and Specific Health Checkups"],"prefix":"10.1007","volume":"38","author":[{"given":"Eri","family":"Hoshino","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Keiko","family":"Konomura","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Masayuki","family":"Obatake","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kensuke","family":"Moriwaki","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michi","family":"Sakai","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kevin Y.","family":"Urayama","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kojiro","family":"Shimozuma","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2022,2,15]]},"reference":[{"issue":"9702","key":"5079_CR1","doi-asserted-by":"publisher","first-page":"1704","DOI":"10.1016\/S0140-6736(09)60946-6","volume":"374","author":"JL Hartley","year":"2009","unstructured":"Hartley JL, Davenport M, Kelly DA (2009) Biliary atresia. Lancet 374(9702):1704\u20131713","journal-title":"Lancet"},{"issue":"6","key":"5079_CR2","doi-asserted-by":"publisher","first-page":"1006","DOI":"10.1016\/S0168-8278(99)80312-2","volume":"31","author":"C Chardot","year":"1999","unstructured":"Chardot C, Carton M, Spire-Bendelac N, Pommelet CL, Golmard J-L, Auvert B (1999) Epidemiology of biliary atresia in France: a national study 1986\u201396. J Hepatol 31(6):1006\u20131013","journal-title":"J Hepatol"},{"issue":"9197","key":"5079_CR3","doi-asserted-by":"publisher","first-page":"25","DOI":"10.1016\/S0140-6736(99)03492-3","volume":"355","author":"PJ McKiernan","year":"2000","unstructured":"McKiernan PJ, Baker AJ, Kelly DA (2000) The frequency and outcome of biliary atresia in the UK and Ireland. Lancet 355(9197):25\u201329","journal-title":"Lancet"},{"issue":"6","key":"5079_CR4","doi-asserted-by":"publisher","first-page":"F451","DOI":"10.1136\/adc.2009.159780","volume":"94","author":"E Livesey","year":"2009","unstructured":"Livesey E, Borja MC, Sharif K et al (2009) Epidemiology of biliary atresia in England and Wales (1999\u20132006). Arch Dis Child Fetal Neonatal Ed 94(6):F451\u2013F455","journal-title":"Arch Dis Child Fetal Neonatal Ed"},{"key":"5079_CR5","doi-asserted-by":"publisher","first-page":"253","DOI":"10.1016\/j.jpeds.2017.05.006","volume":"187","author":"PC Hopkins","year":"2017","unstructured":"Hopkins PC, Yazigi N, Nylund CM (2017) Incidence of biliary atresia and timing of hepatoportoenterostomy in the United States. J Pediatr 187:253\u2013257","journal-title":"J Pediatr"},{"issue":"3","key":"5079_CR6","doi-asserted-by":"publisher","first-page":"299","DOI":"10.1097\/MPG.0b013e3181633562","volume":"46","author":"BE Wildhaber","year":"2008","unstructured":"Wildhaber BE, Majno P, Mayr J et al (2008) Biliary atresia: Swiss national study, 1994\u20132004. J Pediatr Gastroenterol Nutr 46(3):299\u2013307","journal-title":"J Pediatr Gastroenterol Nutr"},{"issue":"8","key":"5079_CR7","doi-asserted-by":"publisher","first-page":"1324","DOI":"10.1016\/j.jpedsurg.2007.03.026","volume":"42","author":"MD Stringer","year":"2007","unstructured":"Stringer MD, Davison SM, Rajwal SR, McClean P (2007) Kasai portoenterostomy: 12-year experience with a novel adjuvant therapy regimen. J Pediatr Surg 42(8):1324\u20131328","journal-title":"J Pediatr Surg"},{"issue":"5","key":"5079_CR8","doi-asserted-by":"publisher","first-page":"1280","DOI":"10.1542\/peds.2008-1949","volume":"123","author":"M-O Serinet","year":"2009","unstructured":"Serinet M-O, Wildhaber BE, Brou\u00e9 P et al (2009) Impact of age at Kasai operation on its results in late childhood and adolescence: a rational basis for biliary atresia screening. Pediatrics 123(5):1280\u20131286","journal-title":"Pediatrics"},{"issue":"6","key":"5079_CR9","doi-asserted-by":"publisher","first-page":"605","DOI":"10.1056\/NEJMc1601230","volume":"375","author":"S Harpavat","year":"2016","unstructured":"Harpavat S, Garcia-Prats JA, Shneider BL (2016) Newborn bilirubin screening for biliary atresia. N Engl J Med 375(6):605\u2013606","journal-title":"N Engl J Med"},{"issue":"1","key":"5079_CR10","doi-asserted-by":"publisher","first-page":"202","DOI":"10.1002\/hep.24023","volume":"53","author":"T-H Lien","year":"2011","unstructured":"Lien T-H, Chang M-H, Wu J-F et al (2011) Effects of the infant stool color card screening program on 5-year outcome of biliary atresia in Taiwan. Hepatology 53(1):202\u2013208","journal-title":"Hepatology"},{"issue":"4","key":"5079_CR11","doi-asserted-by":"publisher","first-page":"1233","DOI":"10.1002\/hep.22182","volume":"47","author":"C-H Hsiao","year":"2008","unstructured":"Hsiao C-H, Chang M-H, Chen H-L et al (2008) Universal screening for biliary atresia using an infant stool color card in Taiwan. Hepatology 47(4):1233\u20131240","journal-title":"Hepatology"},{"issue":"2","key":"5079_CR12","doi-asserted-by":"publisher","first-page":"566","DOI":"10.1002\/hep.21790","volume":"46","author":"RJ Sokol","year":"2007","unstructured":"Sokol RJ, Shepherd RW, Superina R, Bezerra JA, Robuck P, Hoofnagle JH (2007) Screening and outcomes in biliary atresia: summary of a National Institutes of Health workshop. Hepatology 46(2):566\u2013581","journal-title":"Hepatology"},{"issue":"2","key":"5079_CR13","first-page":"219","volume":"56","author":"Japanese Biliary Atresia Society","year":"2020","unstructured":"Japanese Biliary Atresia Society (2020) Summary of Japanese Biliary Atresia Registry 2018. Jpn Soc Pediatr Surg. 56(2):219\u2013225","journal-title":"Jpn Soc Pediatr Surg."},{"issue":"7","key":"5079_CR14","doi-asserted-by":"publisher","first-page":"997","DOI":"10.1016\/S0022-3468(03)00178-7","volume":"38","author":"M Nio","year":"2003","unstructured":"Nio M, Ohi R, Miyano T, Saeki M, Shiraki K, Tanaka K (2003) Five-and 10-year survival rates after surgery for biliary atresia: a report from the Japanese Biliary Atresia Registry. J Pediatr Surg 38(7):997\u20131000","journal-title":"J Pediatr Surg"},{"issue":"3","key":"5079_CR15","doi-asserted-by":"publisher","first-page":"659","DOI":"10.1111\/ajt.14489","volume":"18","author":"M Kasahara","year":"2018","unstructured":"Kasahara M, Umeshita K, Sakamoto S et al (2018) Living donor liver transplantation for biliary atresia: an analysis of 2085 cases in the registry of the Japanese Liver Transplantation Society. Am J Transplant 18(3):659\u2013668","journal-title":"Am J Transplant"},{"issue":"1","key":"5079_CR16","doi-asserted-by":"publisher","first-page":"91","DOI":"10.1097\/MPG.0000000000000569","volume":"60","author":"D Mogul","year":"2015","unstructured":"Mogul D, Zhou M, Intihar P, Schwarz K, Frick K (2015) Cost-effective analysis of screening for biliary atresia with the stool color card. J Pediatr Gastroenterol Nutr 60(1):91\u201398","journal-title":"J Pediatr Gastroenterol Nutr"},{"issue":"3","key":"5079_CR17","doi-asserted-by":"publisher","first-page":"126","DOI":"10.1177\/0969141314542115","volume":"21","author":"RA Schreiber","year":"2014","unstructured":"Schreiber RA, Masucci L, Kaczorowski J et al (2014) Home-based screening for biliary atresia using infant stool colour cards: a large-scale prospective cohort study and cost-effectiveness analysis. J Med Screen 21(3):126\u2013132","journal-title":"J Med Screen"},{"issue":"9796","key":"5079_CR18","doi-asserted-by":"publisher","first-page":"1106","DOI":"10.1016\/S0140-6736(11)60828-3","volume":"378","author":"N Ikegami","year":"2011","unstructured":"Ikegami N, Yoo B-K, Hashimoto H et al (2011) Japanese universal health coverage: evolution, achievements, and challenges. Lancet 378(9796):1106\u20131115","journal-title":"Lancet"},{"key":"5079_CR19","unstructured":"Information Center for Specific Pediatric Chronic Diseases. A Chart for Medical Aid Program for Chronic Pediatric Diseases of Specified Categories. https:\/\/www.shouman.jp\/disease\/. Accessed 11 Jan 2021"},{"key":"5079_CR20","unstructured":"Minds Clinical Guidelines Development Manual 2020 ver.3.0. (2021) https:\/\/minds.jcqhc.or.jp\/s\/manual_2020_3_0. Accessed 4 Jan 2022"},{"issue":"1","key":"5079_CR21","doi-asserted-by":"publisher","first-page":"16","DOI":"10.1186\/s40780-015-0016-5","volume":"1","author":"S Tanaka","year":"2015","unstructured":"Tanaka S, Seto K, Kawakami K (2015) Pharmacoepidemiology in Japan: medical databases and research achievements. J Pharm Health Care Sci 1(1):16","journal-title":"J Pharm Health Care Sci"},{"key":"5079_CR22","volume":"10","author":"K Shinichiro","year":"2018","unstructured":"Shinichiro K, Tatsuya N, Tomoya M et al (2018) National Database of Health Insurance Claims and Specific Health Checkups of Japan (NDB): Outline and Patient-Matching Technique. bioRxiv. 10:280008","journal-title":"bioRxiv."},{"issue":"3","key":"5079_CR23","doi-asserted-by":"publisher","first-page":"485","DOI":"10.1016\/0022-3468(95)90062-4","volume":"30","author":"J Vazquez","year":"1995","unstructured":"Vazquez J, Gutierrez JL, Gamez M et al (1995) Biliary atresia and the polysplenia syndrome: its impact on final outcome. J Pediatr Surg 30(3):485\u2013487","journal-title":"J Pediatr Surg"},{"issue":"3","key":"5079_CR24","doi-asserted-by":"publisher","first-page":"393","DOI":"10.1016\/j.jpeds.2006.05.030","volume":"149","author":"M Davenport","year":"2006","unstructured":"Davenport M, Tizzard SA, Underhill J, Mieli-Vergani G, Portmann B, Had\u017ei\u0107 N (2006) The biliary atresia splenic malformation syndrome: a 28-year single-center retrospective study. J Pediatr 149(3):393\u2013400","journal-title":"J Pediatr"},{"issue":"12","key":"5079_CR25","doi-asserted-by":"publisher","first-page":"2124","DOI":"10.1016\/j.jpedsurg.2015.08.040","volume":"50","author":"M Nio","year":"2015","unstructured":"Nio M, Wada M, Sasaki H, Tanaka H, Watanabe T (2015) Long-term outcomes of biliary atresia with splenic malformation. J Pediatr Surg 50(12):2124\u20132127","journal-title":"J Pediatr Surg"},{"issue":"2","key":"5079_CR26","doi-asserted-by":"publisher","first-page":"57","DOI":"10.15171\/ijhpm.2015.26","volume":"4","author":"N Ikegami","year":"2015","unstructured":"Ikegami N (2015) Fee-for-service payment\u2013an evil practice that must be stamped out? Int J Health Policy Manag 4(2):57","journal-title":"Int J Health Policy Manag"},{"issue":"9","key":"5079_CR27","doi-asserted-by":"publisher","first-page":"630","DOI":"10.1093\/jnci\/djn103","volume":"100","author":"KR Yabroff","year":"2008","unstructured":"Yabroff KR, Lamont EB, Mariotto A et al (2008) Cost of care for elderly cancer patients in the United States. J Natl Cancer Inst 100(9):630\u2013641","journal-title":"J Natl Cancer Inst"},{"key":"5079_CR28","unstructured":"Ministry of Health Labour and Welfare. Revision of medical fees (2021). https:\/\/www.mhlw.go.jp\/stf\/seisakunitsuite\/bunya\/0000106602.html. Accessed 3 Jan 2021"},{"key":"5079_CR29","unstructured":"Ministry of Health Labour and Welfare (2021) Demographic Survey. https:\/\/www.mhlw.go.jp\/toukei\/list\/81-1.html. Accessed 13 Jan 2021"},{"issue":"7","key":"5079_CR30","doi-asserted-by":"publisher","first-page":"1830","DOI":"10.1111\/ajt.12276","volume":"13","author":"M Kasahara","year":"2013","unstructured":"Kasahara M, Umeshita K, Inomata Y, Uemoto S, Society JLT (2013) Long-term outcomes of pediatric living donor liver transplantation in Japan: an analysis of more than 2200 cases listed in the registry of the Japanese Liver Transplantation Society. Am J Transplant 13(7):1830\u20131839","journal-title":"Am J Transplant"},{"key":"5079_CR31","unstructured":"Japan Organ Transplant Network. Procedure Manual for Organ Donation Facilities (2014) https:\/\/www.jotnw.or.jp\/files\/page\/medical\/manual\/doc\/plant.pdf. Accessed 4 Jan 2022"},{"issue":"1","key":"5079_CR32","doi-asserted-by":"publisher","first-page":"66","DOI":"10.1067\/mpd.2001.115068","volume":"139","author":"JC Bucuvalas","year":"2001","unstructured":"Bucuvalas JC, Ryckman FC, Atherton H, Alonso MP, Balistreri WF, Kotagal U (2001) Predictors of cost of liver transplantation in children: a single center study. J Pediatr 139(1):66\u201374","journal-title":"J Pediatr"},{"issue":"5","key":"5079_CR33","doi-asserted-by":"publisher","first-page":"869","DOI":"10.1016\/j.jpedsurg.2006.12.052","volume":"42","author":"G Aspelund","year":"2007","unstructured":"Aspelund G, Ling SC, Ng V, Kim PC (2007) A role for laparoscopic approach in the treatment of biliary atresia and choledochal cysts. J Pediatr Surg 42(5):869\u2013872","journal-title":"J Pediatr Surg"},{"issue":"2","key":"5079_CR34","doi-asserted-by":"publisher","first-page":"203","DOI":"10.1097\/MPG.0000000000001289","volume":"64","author":"S Nightingale","year":"2017","unstructured":"Nightingale S, Stormon MO, O\u2019Loughlin EV et al (2017) Early posthepatoportoenterostomy predictors of native liver survival in biliary atresia. J Pediatr Gastroenterol Nutr 64(2):203\u2013209","journal-title":"J Pediatr Gastroenterol Nutr"},{"key":"5079_CR35","unstructured":"Ando H, Inomata Y, Iwanaka T et al (2020) Clinical practice guidelines for biliary atresia in Japan: a secondary publication of the abbreviated version translated into English. J Hepato Biliary Pancreatic Sci 28(1):55\u201361"}],"container-title":["Pediatric Surgery International"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s00383-022-05079-1.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s00383-022-05079-1\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s00383-022-05079-1.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,3,10]],"date-time":"2022-03-10T12:19:27Z","timestamp":1646914767000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s00383-022-05079-1"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,2,15]]},"references-count":35,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2022,4]]}},"alternative-id":["5079"],"URL":"https:\/\/doi.org\/10.1007\/s00383-022-05079-1","relation":{},"ISSN":["0179-0358","1437-9813"],"issn-type":[{"type":"print","value":"0179-0358"},{"type":"electronic","value":"1437-9813"}],"subject":[],"published":{"date-parts":[[2022,2,15]]},"assertion":[{"value":"27 January 2022","order":1,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"15 February 2022","order":2,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"All authors in this study declared that they do not have anything to disclose regarding conflicts of interest.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}},{"value":"This study was approved by the Institutional Review Board of the National Institute of Public Health (ID#NIPH-IBRA#12306).","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethical approval"}}]}}