The use of simultaneous reprogramming and gene correction to generate an osteogenesis imperfecta patient COL1A1 c. 3936 G>T iPSC line and an isogenic control iPSC line.


Journal

Stem cell research
ISSN: 1876-7753
Titre abrégé: Stem Cell Res
Pays: England
ID NLM: 101316957

Informations de publication

Date de publication:
07 2019
Historique:
received: 17 03 2019
revised: 23 04 2019
accepted: 29 04 2019
pubmed: 15 5 2019
medline: 22 4 2020
entrez: 15 5 2019
Statut: ppublish

Résumé

To develop a disease model for the human 'brittle bone' disease, osteogenesis imperfecta, we used a simultaneous reprogramming and CRISPR-Cas9 genome editing method to produce an iPSC line with the heterozygous patient mutation (COL1A1 c. 3936 G>T) along with an isogenic gene-corrected control iPSC line. Both IPSC lines had a normal karyotype, expressed pluripotency markers and differentiated into cells representative of the three embryonic germ layers. This osteogenesis imperfecta mutant and isogenic iPSC control line will be of use in exploring disease mechanisms and therapeutic approaches in vitro.

Identifiants

pubmed: 31082677
pii: S1873-5061(19)30083-2
doi: 10.1016/j.scr.2019.101453
pii:
doi:

Substances chimiques

Collagen Type I 0
Collagen Type I, alpha 1 Chain 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

101453

Informations de copyright

Copyright © 2019 The Authors. Published by Elsevier B.V. All rights reserved.

Auteurs

Sara Howden (S)

Murdoch Children's Research Institute, Australia.

Hani Hosseini Far (H)

Murdoch Children's Research Institute, Australia; Department of Paediatrics, University of Melbourne, Australia.

Ali Motazedian (A)

Murdoch Children's Research Institute, Australia.

Andrew G Elefanty (AG)

Murdoch Children's Research Institute, Australia; Department of Paediatrics, University of Melbourne, Australia; Department of Anatomy and Developmental Biology, Monash University, Australia.

Edouard G Stanley (EG)

Murdoch Children's Research Institute, Australia; Department of Paediatrics, University of Melbourne, Australia; Department of Anatomy and Developmental Biology, Monash University, Australia.

Shireen R Lamandé (SR)

Murdoch Children's Research Institute, Australia; Department of Paediatrics, University of Melbourne, Australia.

John F Bateman (JF)

Murdoch Children's Research Institute, Australia; Department of Biochemistry and Molecular Biology, University of Melbourne, Australia. Electronic address: john.bateman@mcri.edu.au.

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Classifications MeSH