Province‐wide Biliary Atresia Home Screening Program in British Columbia

ABSTRACT Background and Objectives: Biliary 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...

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Published inJournal of pediatric gastroenterology and nutrition Vol. 66; no. 6; pp. 845 - 849
Main Authors Woolfson, Jessica P., Schreiber, Richard A., Butler, Alison E., MacFarlane, Julie, Kaczorowski, Janusz, Masucci, Lisa, Bryan, Stirling, Collet, Jean Paul
Format Journal Article
LanguageEnglish
Published 01.06.2018
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Abstract ABSTRACT Background and Objectives: Biliary 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. Methods: ISCCs 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. Results: All 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. Conclusions: The 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.
AbstractList ABSTRACT Background and Objectives: Biliary 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. Methods: ISCCs 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. Results: All 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. Conclusions: The 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 Schreiber, Richard A.
MacFarlane, Julie
Bryan, Stirling
Masucci, Lisa
Woolfson, Jessica P.
Collet, Jean Paul
Kaczorowski, Janusz
Butler, Alison E.
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Notes Supplemental digital content is available for this article. Direct URL citations appear in the printed text, and links to the digital files are provided in the HTML text of this article on the journal's Web site
www.jpgn.org
The authors report no conflicts of interest.
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Snippet ABSTRACT Background and Objectives: Biliary atresia (BA), a rare newborn liver disease, is the leading cause of liver‐related death in children. Early disease...
SourceID wiley
SourceType Publisher
StartPage 845
SubjectTerms cost‐effectiveness
neonatal cholestasis
newborn screening
stool color card
Title Province‐wide Biliary Atresia Home Screening Program in British Columbia
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