Krüppeling erythropoiesis: an unexpected broad spectrum of human red blood cell disorders due to KLF1 variants

Blood. 2016 Apr 14;127(15):1856-62. doi: 10.1182/blood-2016-01-694331. Epub 2016 Feb 22.

Abstract

Until recently our approach to analyzing human genetic diseases has been to accurately phenotype patients and sequence the genes known to be associated with those phenotypes; for example, in thalassemia, the globin loci are analyzed. Sequencing has become increasingly accessible, and thus a larger panel of genes can be analyzed and whole exome and/or whole genome sequencing can be used when no variants are found in the candidate genes. By using such approaches in patients with unexplained anemias, we have discovered that a broad range of hitherto unrelated human red cell disorders are caused by variants in KLF1, a master regulator of erythropoiesis, which were previously considered to be extremely rare causes of human genetic disease.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't
  • Review

MeSH terms

  • Anemia, Hemolytic / genetics
  • Animals
  • Blood Group Antigens
  • Erythrocytes / cytology*
  • Erythropoiesis / genetics*
  • Exome
  • Gene Deletion
  • Gene Expression Regulation
  • Genetic Variation
  • Heme / chemistry
  • Hemoglobinopathies / genetics
  • Humans
  • Hydrops Fetalis / genetics
  • Iron / chemistry
  • Kruppel-Like Transcription Factors / genetics*
  • Mice
  • Phenotype
  • Protein Structure, Tertiary
  • Pyruvate Kinase / deficiency
  • Sequence Analysis, DNA
  • beta-Globins / genetics

Substances

  • Blood Group Antigens
  • Kruppel-Like Transcription Factors
  • beta-Globins
  • erythroid Kruppel-like factor
  • Heme
  • Iron
  • Pyruvate Kinase

Associated data

  • PIR/111150
  • PIR/613566
  • PIR/613673