Large deletion of the 5' end of the ROMK1 gene causes antenatal Bartter syndrome

J Am Soc Nephrol. 1998 Dec;9(12):2357-9. doi: 10.1681/ASN.V9122357.

Abstract

Mutations in exon 5 of the ROMK1 gene (KCNJ1) have recently been observed in antenatal Bartter syndrome patients. This study describes a homozygous deletion removing KCNJ1 exons 1 and 2 observed in a consanguineous family with antenatal Bartter syndrome. Absence of the untranslated exon 1 led to the deletion of transcription elements located in this exon that may cause the disease. Deletion of exon 1 transcription elements should lead to the absence of hROMK2-K5 transcripts, whereas hROMK1 transcripts should normally be transcripted. Consequently, probably only hROMK2-K5 transcripts are expressed in the medullary thick ascending limb of Henle.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Bartter Syndrome / embryology
  • Bartter Syndrome / genetics*
  • Consanguinity
  • Exons / genetics
  • Female
  • Fetal Diseases / genetics*
  • Homozygote
  • Humans
  • Infant, Newborn
  • Loop of Henle / metabolism
  • Male
  • Pedigree
  • Polymorphism, Single-Stranded Conformational
  • Potassium Channels / biosynthesis
  • Potassium Channels / genetics*
  • Potassium Channels, Inwardly Rectifying*
  • Protein Isoforms / genetics*
  • RNA, Messenger / biosynthesis
  • RNA, Messenger / genetics
  • Regulatory Sequences, Nucleic Acid
  • Sequence Deletion*
  • Transcription, Genetic

Substances

  • KCNJ1 protein, human
  • Potassium Channels
  • Potassium Channels, Inwardly Rectifying
  • Protein Isoforms
  • RNA, Messenger