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Published on: October 13, 2018
Population-based screening strategies for biliary atresia in the newborn: A systematic review and meta-analysis
Srirupa Hari Gopal1, Rema Zebda1, Arvind Mohan2
1Division of Neonatal-Perinatal Medicine, Baylor College of Medicine & Texas Children's Hospital, Houston, Texas, United States of America.
Insights
Newborn screening for biliary atresia (BA) using direct/conjugated bilirubin (DB/CB) shows high accuracy. Stool color card (SCC) screening is less sensitive but highly specific for early BA detection.
Area of Science:
- Neonatal Medicine
- Pediatric Gastroenterology
- Diagnostic Accuracy Studies
Background:
- Biliary atresia (BA) requires early diagnosis and intervention for improved infant outcomes.
- Newborn screening strategies are being explored to facilitate timely BA detection.
Purpose of the Study:
- To systematically review and assess the diagnostic accuracy of population-based newborn screening strategies for biliary atresia.
- To compare the effectiveness of different screening tests, including stool color card (SCC) and direct/conjugated bilirubin (DB/CB).
Main Methods:
- Systematic review adhering to PRISMA-DTA guidelines, including cohort and cross-sectional studies.
- Meta-analysis of screening tests (SCC, DB/CB) against intraoperative cholangiogram as the reference standard.
- Inclusion of 15 studies encompassing over 1.8 million participants.
Main Results:
- Direct/conjugated bilirubin (DB/CB) assessed post-birth demonstrated high diagnostic accuracy: 100% sensitivity and 98.8% specificity.
- Stool color card (SCC) screening at one month showed 79.6% sensitivity and 99.9% specificity.
- Risk of bias assessment indicated potential concerns regarding patient selection and reference standard in some studies.
Conclusions:
- Direct/conjugated bilirubin (DB/CB) in the initial days of life offers the most accurate diagnostic performance for population-based biliary atresia screening in newborns.
- Future research should investigate the cost-effectiveness and potential benefits of combining screening strategies.
Background:
Newborn screening for biliary atresia (BA) may facilitate earlier diagnosis and intervention for improved clinical outcomes.
Methods:
We systematically reviewed the accuracy of population-based screening strategies for BA in the newborn using PRISMA-DTA guidelines. We included cohort or cross-sectional studies. The screening (index) tests included stool color card (SCC) and direct/conjugated bilirubin (DB/CB) and the reference standard was intraoperative cholangiogram. Meta-analysis was performed using random-effects logistic regression models.
Results:
We included 15 studies (1,816,722 participants) that assessed 5 different population-based screening strategies. QUADAS-2 assessment revealed high risk of bias for patient selection in one study and uncertain risks for reference standard in multiple studies. High certainty evidence suggests that DB/CB assessed after birth had a summary sensitivity of 100% (95% CI 100,100) and specificity of 98.8% (98.8,98.9) (5 studies, 662141 participants). Moderate certainty evidence suggests that SCC screening at a month of age had summary sensitivity of 79.6% (95% CI 70.6, 86.4) and specificity of 99.9% (95% CI 99.9, 99.9) (7 studies, 996262 participants).
Conclusions:
DB/CB in the first few days of life has the best diagnostic accuracy for population screening for biliary atresia in the newborn. Future research should focus on cost-effectiveness and combinations of screening strategies.
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