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Province-wide Biliary Atresia Home Screening Program in British Columbia: Evaluation of First 2 Years
Jessica P Woolfson1,2,3, Richard A Schreiber1,2,3, Alison E Butler1
1Division of Pediatric Gastroenterology, Hepatology and Nutrition.
Insights
Early infant stool color card screening for biliary atresia (BA) in British Columbia showed high specificity and low cost. Successful cases were identified earlier for Kasai hepatoportoenterostomy (KP), improving outcomes for this rare newborn liver disease.
Area of Science:
- Pediatric Gastroenterology
- Neonatal Screening
- Public Health Initiatives
Background:
- Biliary atresia (BA) is a critical neonatal liver disease leading to childhood liver failure.
- Early diagnosis and Kasai hepatoportoenterostomy (KP) surgery improve long-term survival without transplantation.
- Infant stool color cards (ISCCs) have demonstrated effectiveness in universal BA screening programs.
Purpose of the Study:
- To evaluate the performance and cost-effectiveness of a province-wide infant stool color card (ISCC) screening program for biliary atresia (BA) in British Columbia.
- To assess the program's impact on the age of Kasai hepatoportoenterostomy (KP) surgery for identified BA cases.
Main Methods:
- ISCCs were distributed to families at hospital discharge, with instructions for parents to monitor infant stool color.
- Data on live births, ISCC distribution, BA diagnoses, and program costs were collected over a two-year period.
- Program sensitivity, specificity, and predictive values were calculated, alongside the age at KP surgery for confirmed BA cases.
Main Results:
- The program screened 87,583 live births, identifying 6 BA cases with a distribution rate of 94% in participating hospitals.
- Program sensitivity was 50% and specificity was 99%, with a low cost of $0.68 per birth.
- Infants identified through successful program screening had a significantly earlier median age at KP surgery (49 days) compared to those not identified (116 days).
Conclusions:
- The ISCC screening program in British Columbia is characterized by high specificity, broad distribution, and cost-effectiveness.
- Successful identification of BA cases led to earlier surgical intervention (KP), suggesting improved potential for better health outcomes.
- Future program modifications will focus on enhancing sensitivity, with longer-term studies needed to confirm the impact on overall health outcomes.
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.
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