Province-wide Biliary Atresia Home Screening Program in British Columbia: Evaluation of First 2 Years
BACKGROUND AND OBJECTIVESBiliary atresia (BA), a rare newborn liver disease, is the leading cause of liver-related death in children. Early disease recognition and timely surgical Kasai hepatoportoenterostomy (KP) offers long-term survival without liver transplant. Universal BA screening in Taiwan u...
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Published in | Journal of pediatric gastroenterology and nutrition Vol. 66; no. 6; pp. 845 - 849 |
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Main Authors | , , , , , , , |
Format | Journal Article |
Language | English |
Published |
United States
01.06.2018
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Abstract | BACKGROUND AND OBJECTIVESBiliary atresia (BA), a rare newborn liver disease, is the leading cause of liver-related death in children. Early disease recognition and timely surgical Kasai hepatoportoenterostomy (KP) offers long-term survival without liver transplant. Universal BA screening in Taiwan using infant stool color cards (ISCCs) has proven effectiveness. We report our experience with infant stool color card (ISCC) BA screening in a province-wide program in British Columbia (BC). The objective of this study is to assess program performance and cost from launch April 1, 2014 to March 31, 2016. METHODSISCCs distributed to families upon maternity ward discharge. Parents were instructed to monitor their infant's stool color for 1 month and contacted the screening center with concerns. The number of live births, ISCC distribution, BA cases, and costs were recorded. Cases with Program screen success had both acholic stool recognition (ISCC screen success) and timely referral for BA. RESULTSAll 126 maternity units received ISCCs. Of 87,583 live births there were 6 BA cases. Of the 5 cases with ISCC Screen Success 3 had Program Screen Success. The median KP age in the program screen success and failure groups was 49 (42-52) and 116 (49-184) days, respectively. Program sensitivity was 50%, specificity 99%, positive predictive value 4%, and negative predictive value 99%. A random sample of 1054 charts at BC Children's Hospital found an ISCC distribution rate of 94%. After a phase-in period, the annual program cost was $30,033.82, and the ISCC cost per birth was $0.68. CONCLUSIONSThe screening program has high specificity and distribution with low cost. Successful program case identification had earlier age at KP. Program modifications aim to improve sensitivity. Longer-term studies will determine program impact on health outcomes. |
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AbstractList | BACKGROUND AND OBJECTIVESBiliary atresia (BA), a rare newborn liver disease, is the leading cause of liver-related death in children. Early disease recognition and timely surgical Kasai hepatoportoenterostomy (KP) offers long-term survival without liver transplant. Universal BA screening in Taiwan using infant stool color cards (ISCCs) has proven effectiveness. We report our experience with infant stool color card (ISCC) BA screening in a province-wide program in British Columbia (BC). The objective of this study is to assess program performance and cost from launch April 1, 2014 to March 31, 2016. METHODSISCCs distributed to families upon maternity ward discharge. Parents were instructed to monitor their infant's stool color for 1 month and contacted the screening center with concerns. The number of live births, ISCC distribution, BA cases, and costs were recorded. Cases with Program screen success had both acholic stool recognition (ISCC screen success) and timely referral for BA. RESULTSAll 126 maternity units received ISCCs. Of 87,583 live births there were 6 BA cases. Of the 5 cases with ISCC Screen Success 3 had Program Screen Success. The median KP age in the program screen success and failure groups was 49 (42-52) and 116 (49-184) days, respectively. Program sensitivity was 50%, specificity 99%, positive predictive value 4%, and negative predictive value 99%. A random sample of 1054 charts at BC Children's Hospital found an ISCC distribution rate of 94%. After a phase-in period, the annual program cost was $30,033.82, and the ISCC cost per birth was $0.68. CONCLUSIONSThe screening program has high specificity and distribution with low cost. Successful program case identification had earlier age at KP. Program modifications aim to improve sensitivity. Longer-term studies will determine program impact on health outcomes. |
Author | Woolfson, Jessica P Butler, Alison E MacFarlane, Julie Bryan, Stirling Schreiber, Richard A Masucci, Lisa Collet, Jean Paul Kaczorowski, Janusz |
Author_xml | – sequence: 1 givenname: Jessica P surname: Woolfson fullname: Woolfson, Jessica P organization: University of British Columbia – sequence: 2 givenname: Richard A surname: Schreiber fullname: Schreiber, Richard A organization: University of British Columbia – sequence: 3 givenname: Alison E surname: Butler fullname: Butler, Alison E organization: Division of Pediatric Gastroenterology, Hepatology and Nutrition – sequence: 4 givenname: Julie surname: MacFarlane fullname: MacFarlane, Julie organization: Perinatal Services BC, PHSA, Vancouver, BC – sequence: 5 givenname: Janusz surname: Kaczorowski fullname: Kaczorowski, Janusz organization: Dept of Family and Emergency Medicine, Université de Montréal and CRCHUM, Montreal, Quebec – sequence: 6 givenname: Lisa surname: Masucci fullname: Masucci, Lisa organization: St Michaels Hospital, Toronto, Ontario – sequence: 7 givenname: Stirling surname: Bryan fullname: Bryan, Stirling organization: Center for Clinical Epidemiology and Evaluation, Vancouver Coastal Health Research Institute, Vancouver, BC, Canada – sequence: 8 givenname: Jean Paul surname: Collet fullname: Collet, Jean Paul organization: BC Children's Hospital Research Institute, Vancouver, BC, Canada |
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Title | Province-wide Biliary Atresia Home Screening Program in British Columbia: Evaluation of First 2 Years |
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