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

نویسندگان

  • Daisuke Kanda
  • 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
چکیده

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.

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عنوان ژورنال:
  • Drug metabolism and pharmacokinetics

دوره 24 5  شماره 

صفحات  -

تاریخ انتشار 2009