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Neonatal Dubin-Johnson Syndrome and its Differentiation from Biliary Atresia

Journal of Clinical and Translational Hepatology 2023年第1期11卷 163-173页

作者： Teng Liu Jing Zhao Jia-Yan Feng Yi Lu jonathan a.sheps Ren-Xue Wang Jun Han Victor Ling Jian-She Wang The Center for Pediatric Liver Diseases Children’s Hospital of Fudan UniversityShanghaiChina The Department of Pathology Children’s Hospital of Fudan UniversityShanghaiChina BC Cancer Agency VancouverBritish ColumbiaV5Z 1L3Canada University of Victoria-Genome BC Proteomics Center University of VictoriaVictoriaBritish ColumbiaV8Z 7X8Canada Shanghai Key Laboratory of Birth Defects ShanghaiChina

Background and Aims:The aim was to determine if liver biochemistry indices can be used as biomarkers to help differentiate patients with neonatal Dubin–Johnson syndrome(nDJS)from those with biliary atresia(BA).Methods:Patients with genetically-confirmed nDJS or cholangiographically confirmed BA were retrospectively enrolled and randomly assigned to discovery or verification *** liver chemistries,measured during the neonatal period,were *** values were calculated by receiver operating characteristic curve ***:A cohort of 53 nDJS patients was recruited,of whom 13 presented with acholic stools,and 14 underwent diagnostic cholangiography or needle liver biopsy to differentiate from ***-five patients in the cohort,with complete biochemical information measured during the neonatal period,were compared with 133 infants with cholangiographically confirmed *** and direct bilirubin,alanine aminotransferase(ALT),aspartate aminotransferase(AST),total bile acids,alkaline phosphatase,and gamma-glutamyl transferase were significantly lower in nDJS than in *** the discovery cohort,the areas under the curve for ALT and AST were 0.908 and 0.943,*** the validation cohort,13/15 patients in the nDJS group were classified as nDJS,and 10/53 in the BA control group were positive(p<0.00001)with an ALT biomarker cutoff value of 75 IU/*** of 15 patients were classified as nDJS and none were classified positive in the BA group(13/15 vs.0/53,p<0.00001)with an AST cutoff of 87 IU/***:Having assembled and investigated the largest cohort of nDJS patients reported to date,we found that nDJS patients could be distinguished from BA patients using the serum AST level as a *** finding may be clinically useful to spare cholestatic nDJS patients unnecessary invasive procedures.

关键词： Jaundice ABCC2 Cholestasis MRP2 Acholic stools

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