Spatiotemporal cell junction assembly in human iPSC-CM models of arrhythmogenic cardiomyopathy.

Humans Induced Pluripotent Stem Cells beta Catenin / genetics Calcium Signaling Intercellular Junctions Myocytes, Cardiac Plakophilins / genetics
arrhythmogenic cardiomyopathy cardiac tissue engineering cell pairs disease modeling human induced pluripotent stem cells

Journal

Stem cell reports
ISSN: 2213-6711
Titre abrégé: Stem Cell Reports
Pays: United States
ID NLM: 101611300

Informations de publication

Date de publication:
12 09 2023
Historique:
received: 12 01 2023
revised: 21 07 2023
accepted: 22 07 2023
medline: 15 9 2023
pubmed: 19 8 2023
entrez: 18 8 2023
Statut: ppublish

Résumé

Arrhythmogenic cardiomyopathy (ACM) is an inherited cardiac disorder that causes life-threatening arrhythmias and myocardial dysfunction. Pathogenic variants in Plakophilin-2 (PKP2), a desmosome component within specialized cardiac cell junctions, cause the majority of ACM cases. However, the molecular mechanisms by which PKP2 variants induce disease phenotypes remain unclear. Here we built bioengineered platforms using genetically modified human induced pluripotent stem cell-derived cardiomyocytes to model the early spatiotemporal process of cardiomyocyte junction assembly in vitro. Heterozygosity for truncating variant PKP2

Identifiants

DOI: 10.1016/j.stemcr.2023.07.005 PMID: 37595583 PMC: PMC10545490
pubmed: 37595583
pii: S2213-6711(23)00267-9
doi: 10.1016/j.stemcr.2023.07.005
pmc: PMC10545490
pii:
doi:

Substances chimiques

beta Catenin 0
Plakophilins 0

Types de publication

Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, Non-P.H.S.

Langues

eng

Sous-ensembles de citation

IM

Pagination

1811-1826

Subventions

Organisme : NHLBI NIH HHS
ID : R01 HL167450
Pays : United States
Organisme : NHLBI NIH HHS
ID : T32 HL007572
Pays : United States
Organisme : NHLBI NIH HHS
ID : UG3 HL141798
Pays : United States
Organisme : NHLBI NIH HHS
ID : UH3 HL141798
Pays : United States

Informations de copyright

Copyright © 2023 The Author(s). Published by Elsevier Inc. All rights reserved.

Déclaration de conflit d'intérêts

Conflict of interests The authors have no competing interests to disclose.

Références

Biophys Rev. 2018 Aug;10(4):961-971
pubmed: 29876873
Science. 2022 Jul 8;377(6602):180-185
pubmed: 35857545
PLoS Comput Biol. 2011 Feb;7(2):e1001088
pubmed: 21390276
J Cell Biol. 2016 Feb 15;212(4):389-97
pubmed: 26858266
Nat Rev Cardiol. 2019 Sep;16(9):519-537
pubmed: 31028357
Sci Adv. 2021 Oct 15;7(42):eabh3995
pubmed: 34652945
Eur Heart J. 2015 Apr 7;36(14):847-55
pubmed: 25616645
Nat Protoc. 2013 Jan;8(1):162-75
pubmed: 23257984
Circ Res. 2004 Mar 19;94(5):585-91
pubmed: 15031271
Science. 2022 Feb 11;375(6581):639-647
pubmed: 35143298
Mol Cell Biol. 2006 Feb;26(3):898-911
pubmed: 16428445
Stem Cell Reports. 2014 Mar 06;2(3):282-94
pubmed: 24672752
JCI Insight. 2016 Apr 21;1(5):
pubmed: 27170944
Circulation. 2006 Jan 24;113(3):356-64
pubmed: 16415378
Circ Res. 2022 Jun 10;130(12):1780-1802
pubmed: 35679369
Nature. 2013 Feb 7;494(7435):105-10
pubmed: 23354045
Nat Protoc. 2017 Jan;12(1):88-103
pubmed: 27929521
Circ Res. 2009 Sep 11;105(6):523-6
pubmed: 19661460
Cell Commun Adhes. 2014 Jun;21(3):169-79
pubmed: 24766605
Biochim Biophys Acta Mol Basis Dis. 2020 Nov 1;1866(11):165915
pubmed: 32768677
Circulation. 2019 Oct 29;140(18):1491-1505
pubmed: 31533459
J Am Coll Cardiol. 2015 Apr 14;65(14):1438-50
pubmed: 25857910
Sci Transl Med. 2014 Jun 11;6(240):240ra74
pubmed: 24920660
Organogenesis. 2008 Apr;4(2):68-75
pubmed: 19279717
Nat Commun. 2017 Jul 24;8(1):106
pubmed: 28740174
Nat Rev Cardiol. 2018 Jul;15(7):383-384
pubmed: 29795442
N Engl J Med. 2017 Jan 5;376(1):61-72
pubmed: 28052233
Cells. 2021 Aug 14;10(8):
pubmed: 34440861
Stem Cell Reports. 2015 Mar 10;4(3):340-7
pubmed: 25733020
Circulation. 2019 Oct;140(14):1188-1204
pubmed: 31567019
Proc Natl Acad Sci U S A. 2012 Jun 19;109(25):9881-6
pubmed: 22675119
Circulation. 2019 Jul 30;140(5):390-404
pubmed: 31311300
J Cell Sci. 2000 Sep;113 ( Pt 18):3127-39
pubmed: 10954412
Exp Biol Med (Maywood). 2015 Nov;240(11):1543-54
pubmed: 25908635
Circ Res. 2010 Jul 23;107(2):186-99
pubmed: 20651295
Oncogene. 2017 Mar;36(11):1461-1473
pubmed: 27617575
Am J Physiol Heart Circ Physiol. 2012 Jan;302(2):H443-50
pubmed: 22081700
Stem Cell Reports. 2022 Feb 8;17(2):337-351
pubmed: 35063130
J Am Coll Cardiol. 2016 Dec 13;68(23):2540-2550
pubmed: 27931611
Cell Stem Cell. 2020 Jul 2;27(1):50-63.e5
pubmed: 32619518
N Engl J Med. 2009 Mar 12;360(11):1075-84
pubmed: 19279339
Nat Genet. 2004 Nov;36(11):1162-4
pubmed: 15489853
Nat Med. 2014 Jun;20(6):616-23
pubmed: 24813252
Circulation. 1994 Aug;90(2):713-25
pubmed: 8044940
Circ Cardiovasc Genet. 2012 Aug 1;5(4):400-11
pubmed: 22781308
J Clin Invest. 2006 Jul;116(7):2012-21
pubmed: 16823493
Biomaterials. 2010 Jul;31(19):5143-50
pubmed: 20382423
Circ Res. 2007 Sep 28;101(7):703-11
pubmed: 17673670
Sci Transl Med. 2021 Jul 21;13(603):
pubmed: 34290054

Auteurs

Sean L Kim (SL)

Disease Biophysics Group, John A. Paulson School of Engineering and Applied Sciences, Harvard University, Boston, MA 02134, USA; Department of Cardiology, Boston Children's Hospital, Boston, MA 02115, USA.

Michael A Trembley (MA)

Department of Cardiology, Boston Children's Hospital, Boston, MA 02115, USA.

Keel Yong Lee (KY)

Disease Biophysics Group, John A. Paulson School of Engineering and Applied Sciences, Harvard University, Boston, MA 02134, USA; Department of Integrative Bioscience and Biotechnology, Sejong University, Seoul 05006, Republic of Korea.

Suji Choi (S)

Disease Biophysics Group, John A. Paulson School of Engineering and Applied Sciences, Harvard University, Boston, MA 02134, USA.

Luke A MacQueen (LA)

Disease Biophysics Group, John A. Paulson School of Engineering and Applied Sciences, Harvard University, Boston, MA 02134, USA.

John F Zimmerman (JF)

Disease Biophysics Group, John A. Paulson School of Engineering and Applied Sciences, Harvard University, Boston, MA 02134, USA.

Lousanne H C de Wit (LHC)

Disease Biophysics Group, John A. Paulson School of Engineering and Applied Sciences, Harvard University, Boston, MA 02134, USA.

Kevin Shani (K)

Disease Biophysics Group, John A. Paulson School of Engineering and Applied Sciences, Harvard University, Boston, MA 02134, USA; Department of Cardiology, Boston Children's Hospital, Boston, MA 02115, USA.

Douglas E Henze (DE)

Disease Biophysics Group, John A. Paulson School of Engineering and Applied Sciences, Harvard University, Boston, MA 02134, USA.

Daniel J Drennan (DJ)

Disease Biophysics Group, John A. Paulson School of Engineering and Applied Sciences, Harvard University, Boston, MA 02134, USA.

Shaila A Saifee (SA)

Department of Cardiology, Boston Children's Hospital, Boston, MA 02115, USA.

Li Jun Loh (LJ)

Department of Cardiology, Boston Children's Hospital, Boston, MA 02115, USA.

Xujie Liu (X)

Department of Cardiology, Boston Children's Hospital, Boston, MA 02115, USA.

Kevin Kit Parker (KK)

Disease Biophysics Group, John A. Paulson School of Engineering and Applied Sciences, Harvard University, Boston, MA 02134, USA; Department of Cardiology, Boston Children's Hospital, Boston, MA 02115, USA; Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA; Wyss Institute for Biologically Inspired Engineering, Harvard University, Boston, MA 02115, USA. Electronic address: kkparker@g.harvard.edu.

William T Pu (WT)

Department of Cardiology, Boston Children's Hospital, Boston, MA 02115, USA; Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA. Electronic address: william.pu@cardio.chboston.edu.

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Classifications MeSH

questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Primates › Haplorhini › Catarrhini › Hominidae › Humains › Spatiotemporal cell junction assembly in human...
questionsmedicales.fr › Cellules › Cellules souches › Cellules souches pluripotentes › Cellules souches pluripotentes induites › Spatiotemporal cell junction assembly in human...
questionsmedicales.fr › Acides aminés, peptides et protéines › Protéines › Facteurs de transcription › bêta-Caténine › Spatiotemporal cell junction assembly in human...
questionsmedicales.fr › Phénomènes physiologiques cellulaires › Transduction du signal › Systèmes de seconds messagers › Signalisation calcique › Spatiotemporal cell junction assembly in human...
questionsmedicales.fr › Cellules › Structures cellulaires › Membrane cellulaire › Structures de la membrane cellulaire › Jonctions intercellulaires › Spatiotemporal cell junction assembly in human...
questionsmedicales.fr › Cellules › Cellules musculaires › Myocytes cardiaques › Spatiotemporal cell junction assembly in human...
questionsmedicales.fr › Acides aminés, peptides et protéines › Protéines › Protéines du cytosquelette › Plakophilines › Spatiotemporal cell junction assembly in human...