A Highly Phenotyped Open Access Repository of Alpha-1 Antitrypsin Deficiency Pluripotent Stem Cells

Stem Cell Reports. 2020 Jul 14;15(1):242-255. doi: 10.1016/j.stemcr.2020.06.006. Epub 2020 Jul 2.

Abstract

Individuals with the genetic disorder alpha-1 antitrypsin deficiency (AATD) are at risk of developing lung and liver disease. Patient induced pluripotent stem cells (iPSCs) have been found to model features of AATD pathogenesis but only a handful of AATD patient iPSC lines have been published. To capture the significant phenotypic diversity of the patient population, we describe here the establishment and characterization of a curated repository of AATD iPSCs with associated disease-relevant clinical data. To highlight the utility of the repository, we selected a subset of iPSC lines for functional characterization. Selected lines were differentiated to generate both hepatic and lung cell lineages and analyzed by RNA sequencing. In addition, two iPSC lines were targeted using CRISPR/Cas9 editing to accomplish scarless repair. Repository iPSCs are available to investigators for studies of disease pathogenesis and therapeutic discovery.

Keywords: alpha-1 antitrypsin deficiency; hepatic-directed differentiation; iPSC repository; lung progenitor-directed differentiation.

Publication types

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

MeSH terms

  • Access to Information*
  • Adolescent
  • Adult
  • Aged
  • Aged, 80 and over
  • CRISPR-Cas Systems / genetics
  • Cell Differentiation
  • Cell Lineage
  • Databases as Topic*
  • Endoderm / pathology
  • Female
  • Gene Editing
  • Genetic Loci
  • Genotype
  • Hepatocytes / pathology
  • Humans
  • Induced Pluripotent Stem Cells / pathology*
  • Lung / diagnostic imaging
  • Lung / pathology
  • Male
  • Middle Aged
  • Mutation / genetics
  • Phenotype
  • Transcriptome / genetics
  • alpha 1-Antitrypsin / genetics
  • alpha 1-Antitrypsin Deficiency / diagnostic imaging
  • alpha 1-Antitrypsin Deficiency / genetics
  • alpha 1-Antitrypsin Deficiency / pathology*

Substances

  • SERPINA1 protein, human
  • alpha 1-Antitrypsin