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Research article
Development and validation of bile acid profile-based scoring system for identification of biliary atresia: a prospective study
Yongjun Zhang
Shanghai Jiaotong University School of Medicine Xinhua Hospital
Corresponding Author
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Background: Early distinguishing biliary atresia from other causes of infantile cholestasis remains a major challenge. We aimed to develop and validate a scoring system based on bile acid for identification of biliary atresia.
Methods: In a prospective study, a total of 141 infants with cholestasis were enrolled in two sets (derivation cohort, n=66; validation cohort, n=75) from 2014 to 2018. Variables with significant difference between biliary atresia and non-biliary atresia infants were selected in the derivation cohort. Then, a scoring system including those variables was designed and validated.
Results: Among 66 patients in the derivation cohort, 34 (51.5%) had biliary atresia. A scoring system was proposed with the following variables: glycochenodeoxycholic acid/chenodeoxycholic acid, clay stool, and gamma-glutamyl transferase. The total score ranged from 0 to 41, and a cutoff value of 15 identified biliary atresia with an area under receiver operating characteristic curve of 0.87 (95% confidence interval, 0.77–0.94), sensitivity of 85.3%, and specificity of 81.3% in the derivation cohort; these values were also confirmed in a validation cohort with a sensitivity of 90.0% and specificity of 80.0%.
Conclusions: The proposed simple scoring system had good diagnostic accuracy for estimating the risk of biliary atresia in infants with cholestasis.
Keywords
bile acid, biliary atresia, infantile cholestasis, scoring system
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CURRENT STATUS: Under Review
Version 3
Posted 16 May, 2020
No community comments so far
Submission checks complete
On 13 Feb, 2020
No comments provided
Editorial decision: Major revision
On 27 Apr, 2020
Review #2 received
Received 25 Apr, 2020
Review #1 received
Received 21 Apr, 2020
Reviewer #2 agreed
On 08 Apr, 2020
Editor assigned
On 06 Apr, 2020
Reviewers invited
Invitations sent on 06 Apr, 2020
Reviewer #1 agreed
On 06 Apr, 2020
Editor invited
On 05 Apr, 2020
Submission checks complete
On 31 Mar, 2020
No comments provided
Editorial decision: Major revision
On 03 Mar, 2020
Review #2 received
Received 01 Mar, 2020
Review #1 received
Received 01 Mar, 2020
Reviewer #2 agreed
On 16 Feb, 2020
Reviewer #1 agreed
On 15 Feb, 2020
Submission checks complete
On 14 Feb, 2020
Editor assigned
On 14 Feb, 2020
Reviewers invited
Invitations sent on 14 Feb, 2020
Editor invited
On 13 Feb, 2020
First submitted
On 10 Feb, 2020
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This preprint is under consideration at BMC Pediatrics. A preprint is a preliminary version of a manuscript that has not completed peer review at a journal. Research Square does not conduct peer review prior to posting preprints. The posting of a preprint on this server should not be interpreted as an endorsement of its validity or suitability for dissemination as established information or for guiding clinical practice.
Learn more about In Review
Research article
Development and validation of bile acid profile-based scoring system for identification of biliary atresia: a prospective study
Yongjun Zhang
Shanghai Jiaotong University School of Medicine Xinhua Hospital
Corresponding Author
Badges
Prescreen
This manuscript passed our Prescreen assessment
Complete author information
Appropriate declaration statements
Potential risks to human health
Prescreen
Peer Review Timeline
CURRENT STATUS: Under Review
Version 3
Posted 16 May, 2020
No community comments so far
Submission checks complete
On 13 Feb, 2020
No comments provided
Editorial decision: Major revision
On 27 Apr, 2020
Review #2 received
Received 25 Apr, 2020
Review #1 received
Received 21 Apr, 2020
Reviewer #2 agreed
On 08 Apr, 2020
Editor assigned
On 06 Apr, 2020
Reviewers invited
Invitations sent on 06 Apr, 2020
Reviewer #1 agreed
On 06 Apr, 2020
Editor invited
On 05 Apr, 2020
Submission checks complete
On 31 Mar, 2020
No comments provided
Editorial decision: Major revision
On 03 Mar, 2020
Review #2 received
Received 01 Mar, 2020
Review #1 received
Received 01 Mar, 2020
Reviewer #2 agreed
On 16 Feb, 2020
Reviewer #1 agreed
On 15 Feb, 2020
Submission checks complete
On 14 Feb, 2020
Editor assigned
On 14 Feb, 2020
Reviewers invited
Invitations sent on 14 Feb, 2020
Editor invited
On 13 Feb, 2020
First submitted
On 10 Feb, 2020
Metrics
Comments: 0
PDF Downloads: ...
HTML Views: ...
License:
This work is licensed under a CC BY 4.0 License. Read Full License
Background: Early distinguishing biliary atresia from other causes of infantile cholestasis remains a major challenge. We aimed to develop and validate a scoring system based on bile acid for identification of biliary atresia.
Methods: In a prospective study, a total of 141 infants with cholestasis were enrolled in two sets (derivation cohort, n=66; validation cohort, n=75) from 2014 to 2018. Variables with significant difference between biliary atresia and non-biliary atresia infants were selected in the derivation cohort. Then, a scoring system including those variables was designed and validated.
Results: Among 66 patients in the derivation cohort, 34 (51.5%) had biliary atresia. A scoring system was proposed with the following variables: glycochenodeoxycholic acid/chenodeoxycholic acid, clay stool, and gamma-glutamyl transferase. The total score ranged from 0 to 41, and a cutoff value of 15 identified biliary atresia with an area under receiver operating characteristic curve of 0.87 (95% confidence interval, 0.77–0.94), sensitivity of 85.3%, and specificity of 81.3% in the derivation cohort; these values were also confirmed in a validation cohort with a sensitivity of 90.0% and specificity of 80.0%.
Conclusions: The proposed simple scoring system had good diagnostic accuracy for estimating the risk of biliary atresia in infants with cholestasis.
Figure 1
Figure 2