Literature DB >> 32436673

Neonatal cholestasis due to citrin deficiency: diagnostic pitfalls.

Patryk Lipiński1, Dorota Jurkiewicz2, Elżbieta Ciara2, Rafał Płoski3, Sabina Więcek4, Anna Bogdańska5, Teresa Stradomska5, Piotr Socha6, Dariusz Rokicki7, Anna Tylki-Szymańska1, Irena Jankowska6.   

Abstract

Citrin deficiency can manifest in newborns or infants as neonatal intrahepatic cholestasis caused by citrin deficiency (NICCD). The paper presents a case of Polish NICCD patient presenting with low birth weight, failure to thrive, prolonged cholestatic jaundice with coagulopathy and hypoalbuminemia with normal results of MS/MS newborn screening but with high blood citrulline level observed at 3 months of age. Unreported findings included N-hypoglycosylation and increased serum very-long-chain fatty acids (VLCFA), probably secondary to liver impairment. Final diagnosis was established based on whole-exome sequencing (WES) analysis.

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Year:  2020        PMID: 32436673     DOI: 10.18388/abp.2020_5202

Source DB:  PubMed          Journal:  Acta Biochim Pol        ISSN: 0001-527X            Impact factor:   2.349


  1 in total

1.  [Clinical characteristics and gene variants of patients with infantile intrahepatic cholestasis].

Authors:  Mei-Juan Wang; Xue-Mei Zhong; Xin Ma; Hui-Juan Ning; Dan Zhu; You-Zhe Gong; Meng Jin
Journal:  Zhongguo Dang Dai Er Ke Za Zhi       Date:  2021-01
  1 in total

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