Literature DB >> 19881259

Novel large-scale deletion (whole exon 7) in the ABCC2 gene in a patient with the Dubin-Johnson syndrome.

Daisuke Kanda1, Hitoshi Takagi, Yasutsugu Kawahara, Yutaka Yata, Tomofumi Takakusagi, Takeshi Hatanaka, Teruo Yoshinaga, Keigo Iesaki, Kenji Kashiwabara, Tsugio Higuchi, Masatomo Mori, Takeshi Hirota, Shun Higuchi, Ichiro Ieiri.   

Abstract

The Dubin-Johnson syndrome (DJS) is an inherited liver disorder characterized by conjugated hyperbilirubinemia and caused by ABCC2 gene mutations resulting in deficiency of multidrug resistance associated-protein 2 (MRP2) function. A 76-year-old woman with serious jaundice was referred to our hospital. She was clinically diagnosed with DJS with hepatic congestion, due to constrictive pericarditis. We analyzed all exons and exon-intron junctions of the ABCC2 gene by DNA sequencing and identified a new large-scale deletion, 1008 bp, including the whole exon 7, as homozygosity. Some mutations in the ABCC2 gene associated with splicing errors have been reported in intronic regions; however, this is a new type of large-scale deletion detectable in the genomic DNA sequence. Severe hyperbilirubinemia is rare in patients with constrictive pericarditis and this case suggests that MRP2 may play a crucial role in compensating for the serum bilirubin in congestive hepatopathy.

Entities:  

Mesh:

Substances:

Year:  2009        PMID: 19881259     DOI: 10.2133/dmpk.24.464

Source DB:  PubMed          Journal:  Drug Metab Pharmacokinet        ISSN: 1347-4367            Impact factor:   3.614


  6 in total

1.  In Vitro Transport Activity and Trafficking of MRP2/ABCC2 Polymorphic Variants.

Authors:  Xia Wen; Melanie S Joy; Lauren M Aleksunes
Journal:  Pharm Res       Date:  2017-04-12       Impact factor: 4.200

Review 2.  Alternative Splicing: Expanding Diversity in Major ABC and SLC Drug Transporters.

Authors:  Ji Eun Park; Gongmi Ryoo; Wooin Lee
Journal:  AAPS J       Date:  2017-10-02       Impact factor: 4.009

Review 3.  Multidrug resistance proteins (MRPs/ABCCs) in cancer chemotherapy and genetic diseases.

Authors:  Zhe-Sheng Chen; Amit K Tiwari
Journal:  FEBS J       Date:  2011-08-01       Impact factor: 5.542

4.  Case Report: Dubin-Johnson Syndrome Presenting With Infantile Cholestasis: An Overlooked Diagnosis in an Extended Family.

Authors:  Naglaa M Kamal; Omar Saadah; Hamdan Alghamdi; Ali Algarni; Mortada H F El-Shabrawi; Laila M Sherief; Salma A S Abosabie
Journal:  Front Pediatr       Date:  2022-05-25       Impact factor: 3.569

5.  eUnaG: a new ligand-inducible fluorescent reporter to detect drug transporter activity in live cells.

Authors:  Johannes T-H Yeh; Kwangho Nam; Joshua T-H Yeh; Norbert Perrimon
Journal:  Sci Rep       Date:  2017-02-08       Impact factor: 4.379

Review 6.  Membrane transporters in a human genome-scale metabolic knowledgebase and their implications for disease.

Authors:  Swagatika Sahoo; Maike K Aurich; Jon J Jonsson; Ines Thiele
Journal:  Front Physiol       Date:  2014-03-11       Impact factor: 4.566

  6 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.