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Maternal risk factors associated with offspring biliary atresia: population-based study



Biliary atresia (BA) is a progressive, idiopathic, fibro-obliterative disease of the intra and extrahepatic biliary tree. If untreated, it results in severe liver injury and death. The etiology and pathogenesis of BA remain unclear. Few studies have investigated the association between maternal illness/drug use and the occurrence of BA in offspring.


We used the data from the Birth Certificate Application of Taiwan and linked to National Health Insurance Research Database and Taiwan Maternal and Child Health Database for the years 2004 to 2017 (N = 1,647,231) on 2022/03, and identified BA cases according to diagnosis and procedure code. A total of 285 BA cases were identified.


Mothers with type 2 diabetes mellitus and non-dependent drug abuse had higher rates having BA children than non-BA children, with an odds ratio of 2.17 (95% confidence interval [CI] = 1.04–4.53) and OR: 3.02 (95% CI = 1.34–6.78), respectively.


These results support the notion that BA occurrence is related to maternal reasons. Further studies should be designed to identify additional maternal and pregnancy risk factors and to understand the underlying pathophysiology.


  1. 1.

    The occurrence of offspring biliary atresia may be related to maternal illness/drug use.

  2. 2.

    Maternal drug abuse and type 2 diabetes mellitus pose a high risk for offspring biliary atresia.

  3. 3.

    If maternal etiology is found, biliary atresia might be a preventable disease.

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Fig. 1

Data availability

The data underlying this study is from the National Health Insurance Research Database, which has been transferred to the Health and Welfare Data Science Center (HWDC). Requests for data can be sent as a formal proposal to the HWDC, Department of Statistics, Ministry of Health and Welfare, Taiwan ( with an IRB approval for research purpose only. To access the data, please contact the HWDC (;


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The authors would like to thank the Health Information and Epidemiology Laboratory (CLRPG6L0041) at Chiayi Chang Gung Memorial Hospital for their comments and assistance in the data analysis.


This study was supported in part by grants CFRPG6K0041 (J. M. Sheen) from Chiayi Chang Gung Memorial Hospital, Taiwan.

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Authors and Affiliations



C.C.M. and K.K.C. wrote the first draft of the manuscript. C.W.H., and S.C.H., reviewed and edited the subsequent drafts. L.C.P and C.K.J. performed formal analyses. Y.Y.H validated the statistical analyses. Y.J.B. and S.J.M. supervised and critically reviewed the manuscript. All authors approved the final version of the manuscript.

Corresponding authors

Correspondence to Ju-Bei Yen or Jiunn-Ming Sheen.

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Because this was a secondary data analysis, all identifications of patients and institutions were removed before data release, so patient consent was not required.

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The authors declare no competing interests.

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Chang, CM., Kuo, KC., Chen, WH. et al. Maternal risk factors associated with offspring biliary atresia: population-based study. Pediatr Res (2022).

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