The Epidemiology of Biliary Atresia: Exploring the Role of Developmental Factors on Birth Prevalence

Cavallo, Laurel; Kovar, Erin; Aqul, Amal; McLoughlin, Lucille; Mittal, Naveen K; Rodriguez‐Baez, Norberto; Shneider, Benjamin L.; Zwiener, Robert J.; Chambers, Tiffany M.; Langlois, Peter H.; Canfield, Mark A.; Agopian, A. J.; Lupo, Philip J.; Harpavat, Sanjiv

doi:10.1016/j.jpeds.2022.03.038

Cited by 16 publications

(8 citation statements)

References 27 publications

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“…The results showed that prematurity was associated with BA, which is consistent with findings of previous reports . One possible assumption is that an intrauterine event, such as maternal infection, exerts stress on the fetus, leads to preterm birth, and is associated with the development of BA.…”

Section: Discussionsupporting

confidence: 91%

“…We used the annual insurance premium as a proxy for socioeconomic level because Taiwan’s National Health Insurance program operates under a progressive insurance rate system; that is, people with higher income pay higher annual insurance premiums . We also obtained additional perinatal confounders of BA and maternal infection: female sex, preterm labor (childbirth before 37 weeks’ gestation), infant birth weight, preeclampsia, and maternal diabetes …”

Section: Methodsmentioning

confidence: 99%

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Maternal Prenatal Infections and Biliary Atresia in Offspring

Wang,

Chiu,

Kuo

et al. 2024

JAMA Netw Open

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ImportanceInvestigations into the association of antepartum maternal infections with the pathogenesis of biliary atresia (BA) in human offspring are insufficient.ObjectiveTo examine the association between prenatal infections in mothers and the development of BA in their offspring.Design, Setting, and ParticipantsThis population-based case-control study obtained administrative data from the Taiwan National Health Insurance Research Database with linkage to the Taiwan Maternal and Child Health Database, capturing demographic and medical information on nearly all 23 million of the Taiwan population. The cohort comprised 2 905 978 singleton live births among mother-infant dyads between January 1, 2004, and December 31, 2020, in Taiwan. The case group of infants with BA was identified from use of International Classification of Diseases diagnostic codes for BA and subsequent Kasai procedure or liver transplant. The control group was randomly selected from infants without BA, representing approximately 1 in 1000 study population. Data analyses were performed from May 1 to October 31, 2023.ExposurePrenatal maternal infections, including intestinal infection, influenza, upper airway infection, pneumonia, soft-tissue infection, and genitourinary tract infection.Main Outcomes and MeasuresThe main outcome was exposure to prenatal maternal infections. Inverse probability weighting analysis was performed by building a logistic regression model to estimate the probability of the exposure observed for a particular infant and using the estimated probability as a weight in subsequent analyses. The weighted odds ratio (OR) estimated by logistic regressions was then used to assess the risk of BA in offspring after prenatal maternal infections.ResultsAmong the mother-infant dyads included, 447 infants with BA were cases (232 females [51.9%]) and 2912 infants without BA were controls (1514 males [52.0%]). The mean (SD) maternal age at childbirth was 30.7 (4.9) years. Offspring exposed to prenatal intestinal infection (weighted OR, 1.46; 95% CI, 1.17-1.82) and genitourinary tract infection (weighted OR, 1.22; 95% CI, 1.05-1.41) in mothers exhibited a significantly higher risk of BA. Furthermore, maternal intestinal infection (weighted OR, 6.05; 95% CI, 3.80-9.63) and genitourinary tract infection (weighted OR, 1.55; 95% CI, 1.13-2.11) that occurred during the third trimester were associated with an increased risk of BA in offspring.Conclusions and RelevanceResults of this case-control study indicate an association between prenatal intestinal infection and genitourinary tract infection in mothers and BA occurrence in their offspring. Further studies are warranted to explore the underlying mechanisms of this association.

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Section: Discussionsupporting

confidence: 91%

Section: Methodsmentioning

confidence: 99%

Maternal Prenatal Infections and Biliary Atresia in Offspring

Wang,

Chiu,

Kuo

et al. 2024

JAMA Netw Open

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“…We evaluated whether an association between gestational K. brevis exposure and BA exists and observed no significant association using these data. The incidence of BA is increasing over time, with conflicting data regarding seasonality and risk factors 35,36 . Our study did not account for the many proposed, yet not definitive, genetic, environmental or infectious confounders.…”

Section: Discussionmentioning

confidence: 99%

“…Interestingly, at a 50-mile radius, the median gestational exposure for cases peaked at 6 months while for controls the peak occurred at 9 months. Recent studies explore the influence of early in utero events and the development of BA 36 , and so it is worth noting that the timing of a toxic insult may be more important than absolute exposure.…”

Section: K Brevis Levels Can Fluctuate Daily Depending On Various Met...mentioning

confidence: 99%

A Case-Control Study of the Association BetweenKarenia Brevis(Red Tide) and Biliary Atresia

Wyrebek

Fierstein

Wells

et al. 2022

Preprint

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ObjectiveThe study objective was to evaluate the association between maternal Karenia brevis (K. brevis) exposure during pregnancy and the prevalence of biliary atresia (BA) in offspring.Study DesignThis was a hospital-based, case-control study in which cases were infants diagnosed with BA at Johns Hopkins All Childrens Hospital from October 2001 to December 2019. Controls were matched 4:1 by age, randomly selected from healthy infants hospitalized during the study period for common pediatric diagnoses. Infants were excluded if they had congenital anomalies and/or were non-Florida residents. Gestational K. brevis exposure levels (cells/liter) were determined from Florida Fish and Wildlife Conservation Commission exposure data at 10- and 50-mile radii from the maternal zip code of residence. Multivariable conditional logistic regression determined adjusted odds of BA in offspring based on maternal gestational K. brevis exposure.ResultsOf 38 cases and 152 controls, no significant inter-group differences were observed in race/ethnicity, season of birth or coastal residence. Median gestational exposure at the 10-mile radius was 0 cells/liter in both groups. A greater proportion of cases had no K. brevis exposure (63.2%, n = 24) in comparison to controls (37.5%, n = 57; p = .04) at a 10-mile radius. At the 50-mile radius, cases had a peak median exposure at 6 months of gestation compared to controls peak at 9 months. After adjustment for sex, seasonality, race/ethnicity, and coastal residence, there was no significant association between BA and maximum K. brevis exposure per trimester of pregnancy at the 10- or 50-mile radius.ConclusionWe observed no association between gestational K. brevis (cells/liter) exposure at a 10- and 50-mile radius from maternal zip code of residence and BA in offspring.

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“…The prevalence of congenital biliary atresia varies by geography and ethnicity There are significant regional differences in the incidence of BA, ranging from 1:15,000 to 19,000 in Europe and 1:5–10 000 in Japan and China. In addition, both intrahepatic and extrahepatic bile duct obstruction can lead to biliary atresia, but extrahepatic obstruction is typical ( Mack and Sokol, 2005 ; Cavallo et al, 2022 ).…”

Section: Biliary Atresiamentioning

confidence: 99%

Genetic alterations and molecular mechanisms underlying hereditary intrahepatic cholestasis

Xie

Wei

et al. 2023

Front. Pharmacol.

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Hereditary cholestatic liver disease caused by a class of autosomal gene mutations results in jaundice, which involves the abnormality of the synthesis, secretion, and other disorders of bile acids metabolism. Due to the existence of a variety of gene mutations, the clinical manifestations of children are also diverse. There is no unified standard for diagnosis and single detection method, which seriously hinders the development of clinical treatment. Therefore, the mutated genes of hereditary intrahepatic cholestasis were systematically described in this review.

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The Epidemiology of Biliary Atresia: Exploring the Role of Developmental Factors on Birth Prevalence

Cited by 16 publications

References 27 publications

Maternal Prenatal Infections and Biliary Atresia in Offspring

Maternal Prenatal Infections and Biliary Atresia in Offspring

A Case-Control Study of the Association BetweenKarenia Brevis(Red Tide) and Biliary Atresia

Genetic alterations and molecular mechanisms underlying hereditary intrahepatic cholestasis

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