Induced pluripotent stem cells: a novel frontier in the study of human primary immunodeficiencies

J Allergy Clin Immunol. 2011 Jun;127(6):1400-7.e4. doi: 10.1016/j.jaci.2010.11.008. Epub 2010 Dec 24.

Abstract

Background: The novel ability to epigenetically reprogram somatic cells into induced pluripotent stem cells (iPSCs) through the exogenous expression of transcription promises to revolutionize the study of human diseases.

Objective: Here we report on the generation of 25 iPSC lines from 6 patients with various forms of primary immunodeficiencies (PIDs) affecting adaptive immunity, innate immunity, or both.

Methods: Patients' dermal fibroblasts were reprogrammed by expression of 4 transcription factors, octamer-binding transcription factor 4 (OCT4), sex determining region Y-box 2 (SOX2), Krueppel-like factor 4 (KLF4), and cellular myelomonocytosis proto-oncogene (cMYC), by using a single excisable polycistronic lentiviral vector.

Results: iPSCs derived from patients with PIDs show a stemness profile that is comparable with that observed in human embryonic stem cells. After in vitro differentiation into embryoid bodies, pluripotency of the patient-derived iPSC lines was demonstrated by expression of genes characteristic of each of the 3 embryonic layers. We have confirmed the patient-specific origin of the iPSC lines and ascertained maintenance of karyotypic integrity.

Conclusion: By providing a limitless source of diseased stem cells that can be differentiated into various cell types in vitro, the repository of iPSC lines from patients with PIDs represents a unique resource to investigate the pathophysiology of hematopoietic and extrahematopoietic manifestations of these diseases and might assist in the development of novel therapeutic approaches based on gene correction.

Publication types

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

MeSH terms

  • Adaptive Immunity
  • Cell Dedifferentiation
  • Cell Differentiation
  • Cell Line
  • Cell Transdifferentiation
  • DNA / genetics
  • Gene Expression
  • Genes, myc
  • Humans
  • Immunity, Innate
  • Immunologic Deficiency Syndromes / genetics
  • Immunologic Deficiency Syndromes / pathology*
  • Immunologic Deficiency Syndromes / physiopathology*
  • Induced Pluripotent Stem Cells / pathology*
  • Induced Pluripotent Stem Cells / physiology*
  • Karyotyping
  • Kruppel-Like Factor 4
  • Kruppel-Like Transcription Factors / genetics
  • Octamer Transcription Factor-3 / genetics
  • Proto-Oncogene Mas
  • SOXB1 Transcription Factors / genetics

Substances

  • KLF4 protein, human
  • Kruppel-Like Factor 4
  • Kruppel-Like Transcription Factors
  • MAS1 protein, human
  • Octamer Transcription Factor-3
  • POU5F1 protein, human
  • Proto-Oncogene Mas
  • SOX2 protein, human
  • SOXB1 Transcription Factors
  • DNA