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Alagille Syndrome Mimicking Biliary Atresia in Early Infancy

The result's identifiers

  • Result code in IS VaVaI

    <a href="https://www.isvavai.cz/riv?ss=detail&h=RIV%2F00216208%3A11130%2F15%3A10315602" target="_blank" >RIV/00216208:11130/15:10315602 - isvavai.cz</a>

  • Alternative codes found

    RIV/00064203:_____/15:10315602 RIV/00023001:_____/15:00059573

  • Result on the web

    <a href="http://dx.doi.org/10.1371/journal.pone.0143939" target="_blank" >http://dx.doi.org/10.1371/journal.pone.0143939</a>

  • DOI - Digital Object Identifier

    <a href="http://dx.doi.org/10.1371/journal.pone.0143939" target="_blank" >10.1371/journal.pone.0143939</a>

Alternative languages

  • Result language

    angličtina

  • Original language name

    Alagille Syndrome Mimicking Biliary Atresia in Early Infancy

  • Original language description

    Alagille syndrome may mimic biliary atresia in early infancy. Since mutations in JAG1 typical for Alagille syndrome type 1 have also been found in biliary atresia, we aimed to identify JAG1 mutations in newborns with proven biliary atresia (n = 72). Fivebiliary atresia patients with cholestasis, one additional characteristic feature of Alagille syndrome and ambiguous liver histology were single heterozygotes for nonsense or frameshift mutations in JAG1. No mutations were found in the remaining 67 patients. All "biliary atresia" carriers of JAG1 null mutations developed typical Alagille syndrome at the age of three years. Our data do not support association of biliary atresia with JAG1 mutations, at least in Czech patients. Rapid testing for JAG1 mutations could prevent misdiagnosis of Alagille syndrome in early infancy and improve their outcome.

  • Czech name

  • Czech description

Classification

  • Type

    J<sub>x</sub> - Unclassified - Peer-reviewed scientific article (Jimp, Jsc and Jost)

  • CEP classification

    FG - Paediatrics

  • OECD FORD branch

Result continuities

  • Project

  • Continuities

    S - Specificky vyzkum na vysokych skolach<br>I - Institucionalni podpora na dlouhodoby koncepcni rozvoj vyzkumne organizace

Others

  • Publication year

    2015

  • Confidentiality

    S - Úplné a pravdivé údaje o projektu nepodléhají ochraně podle zvláštních právních předpisů

Data specific for result type

  • Name of the periodical

    PLoS ONE

  • ISSN

    1932-6203

  • e-ISSN

  • Volume of the periodical

    10

  • Issue of the periodical within the volume

    11

  • Country of publishing house

    US - UNITED STATES

  • Number of pages

    7

  • Pages from-to

  • UT code for WoS article

    000365889800091

  • EID of the result in the Scopus database

    2-s2.0-84957105474