A Human iPS Cell-Derived Artificial Skeletal Muscle for Regenerative Medicine, Disease Modelling and Drug Screening

Project Information

HISTOID

Grant agreement ID: 759108

DOI

10.3030/759108

Project closed

EC signature date 7 August 2018

Start date 1 September 2018

End date 28 February 2025

Funded under

• EXCELLENT SCIENCE - European Research Council (ERC)

Total cost

€ 1 499 738,00

EU contribution

€ 1 499 738,00

Coordinated by

UNIVERSITY COLLEGE LONDON
United Kingdom

CORDIS provides links to public deliverables and publications of HORIZON projects.

Links to deliverables and publications from FP7 projects, as well as links to some specific result types such as dataset and software, are dynamically retrieved from OpenAIRE .

Publications

Other (3)

DLL4 and PDGF-BB regulate migration of human iPSC-derived skeletal myogenic progenitors

Author(s): Giulia Ferrari, SungWoo Choi, Louise A. Moyle, Mackinlay, Naira Naouar, Christine Wells, Francesco Muntoni, Francesco Saverio Tedesco
Published in: Biorxiv, 2021
Publisher: Cold Spring Harbor Laboratory
DOI: 10.1101/2021.02.28.431778

CRISPR-Cas9 mediated endogenous utrophin upregulation improves Duchenne Muscular Dystrophy

Author(s): Simon Guiraud, Sumitava Dastidar, Fetta Mazed, Fatima Amor, Maelle Ralu, Anne de Cian, Isabelle Richard, Giuseppe Ronzitti, Francesco Saverio Tedesco, Mario Amendola
Published in: 2023
Publisher: Cold Spring Harbor Laboratory
DOI: 10.1101/2023.04.18.536394

Advanced human iPSC-based modelling of <i>LMNA</i> -related congenital muscular dystrophy enables development of targeted genetic therapies for muscle laminopathies

Author(s): Daniel P. Moore, Heather B. Steele-Stallard, Luca Pinton, Valentina Maria Lionello, Lucia Rossi, Artadokht Aghaeipour, Salma Jalal, Cherry Tsz Yan Wong, Angela Clara-Hwang, Gisèle Bonne, Peter S. Zammit, Francesco Saverio Tedesco
Published in: BioRxiv, 2025
Publisher: Cold Spring Harbor Laboratory
DOI: 10.1101/2025.06.22.660928

Peer reviewed articles (16)

3D human induced pluripotent stem cell–derived bioengineered skeletal muscles for tissue, disease and therapy modeling

Author(s): Luca Pinton, Moustafa Khedr, Valentina M. Lionello, Shilpita Sarcar, Sara M. Maffioletti, Sumitava Dastidar, Elisa Negroni, SungWoo Choi, Noreen Khokhar, Anne Bigot, John R. Counsell, Andreia Sofia Bernardo, Peter S. Zammit, Francesco Saverio Tedesco
Published in: Nature Protocols, Issue 18, 2023, Page(s) 1337-1376, ISSN 1754-2189
Publisher: Nature Publishing Group
DOI: 10.1038/s41596-022-00790-8

Metabolic remodeling in hiPSC-derived myofibers carrying the m.3243A&gt;G mutation

Author(s): Gabriel E. Valdebenito, Anitta R. Chacko, Chih-Yao Chung, Preethi Sheshadri, Haoyu Chi, Benjamin O'Callaghan, Monika J. Madej, Henry Houlden, Hannah Rouse, Valle Morales, Katiuscia Bianchi, Francesco Saverio Tedesco, Robert D.S. Pitceathly, Michael R. Duchen
Published in: Stem Cell Reports, Issue 20, 2025, Page(s) 102448, ISSN 2213-6711
Publisher: Cell Press
DOI: 10.1016/j.stemcr.2025.102448

Assessing and enhancing migration of human myogenic progenitors using directed <scp>iPS</scp> cell differentiation and advanced tissue modelling

Author(s): SungWoo Choi, Giulia Ferrari, Louise A Moyle, Kirsty Mackinlay, Naira Naouar, Salma Jalal, Sara Benedetti, Christine Wells, Francesco Muntoni, Francesco Saverio Tedesco
Published in: EMBO Molecular Medicine, Issue 14, 2023, ISSN 1757-4676
Publisher: John Wiley & Sons Ltd.
DOI: 10.15252/emmm.202114526

Generation of a human induced pluripotent stem cell line (CRICKi021-A) from a patient with Ullrich congenital muscular dystrophy carrying a pathogenic mutation in the COL6A1 gene

Author(s): Yunsong Jiang, Liani G. Devito, Francesco Muntoni, Lyn Healy, Francesco Saverio Tedesco
Published in: Stem Cell Research, Issue 83, 2025, Page(s) 103648, ISSN 1873-5061
Publisher: Elsevier
DOI: 10.1016/j.scr.2024.103648

Engineering of human induced pluripotent stem cells via human artificial chromosome vectors for cell therapy and disease modeling

Author(s): Yasuhiro Kazuki, Narumi Uno, Satoshi Abe, Naoyo Kajitani, Kanako Kazuki, Yuwna Yakura, Chiaki Sawada, Shuta Takata, Masaki Sugawara, Yuichi Nagashima, Akane Okada, Masaharu Hiratsuka, Mitsuhiko Osaki, Giulia Ferrari, Francesco Saverio Tedesco, Satoshi Nishikawa, Ken Fukumoto, Shin-ichiro Takayanagi, Atsushi Kunisato, Shin Kaneko, Mitsuo Oshimura, Kazuma Tomizuka
Published in: Molecular Therapy - Nucleic Acids, Issue 23, 2021, Page(s) 629-639, ISSN 2162-2531
Publisher: Nature Publishing Group
DOI: 10.1016/j.omtn.2020.12.012

Bioengineering human skeletal muscle models: Recent advances, current challenges and future perspectives

Author(s): Yunsong Jiang, Tugce Torun, Sara M. Maffioletti, Andrea Serio, Francesco Saverio Tedesco
Published in: Experimental Cell Research, Issue 416, 2024, Page(s) 113133, ISSN 0014-4827
Publisher: Academic Press
DOI: 10.1016/j.yexcr.2022.113133

Cellular dynamics of myogenic cell migration: molecular mechanisms and implications for skeletal muscle cell therapies

Author(s): SungWoo Choi, Giulia Ferrari, Francesco Saverio Tedesco
Published in: EMBO Molecular Medicine, Issue 12/12, 2020, ISSN 1757-4676
Publisher: John Wiley & Sons Ltd.
DOI: 10.15252/emmm.202012357

Generation of an MTM1-mutant iPSC line (CRICKi008-A) from an individual with X-linked myotubular myopathy (XLMTM)

Author(s): Liani G. Devito, Valentina M. Lionello, Francesco Muntoni, Francesco Saverio Tedesco, Lyn Healy
Published in: Stem Cell Research, Issue 69, 2024, Page(s) 103079, ISSN 1873-5061
Publisher: Elsevier
DOI: 10.1016/j.scr.2023.103079

Modeling Skeletal Muscle Laminopathies Using Human Induced Pluripotent Stem Cells Carrying Pathogenic LMNA Mutations

Author(s): Heather B. Steele-Stallard, Luca Pinton, Shilpita Sarcar, Tanel Ozdemir, Sara M. Maffioletti, Peter S. Zammit, Francesco Saverio Tedesco
Published in: Frontiers in Physiology, Issue 9, 2018, ISSN 1664-042X
Publisher: Frontiers Research Foundation
DOI: 10.3389/fphys.2018.01332

Low-cost, versatile, and highly reproducible microfabrication pipeline to generate 3D-printed customised cell culture devices with complex designs

Author(s): Cathleen Hagemann, Matthew C. D. Bailey, Eugenia Carraro, Ksenia S. Stankevich, Valentina Maria Lionello, Noreen Khokhar, Pacharaporn Suklai, Carmen Moreno-Gonzalez, Kelly O’Toole, George Konstantinou, Christina L. Dix, Sudeep Joshi, Eleonora Giagnorio, Mads S. Bergholt, Christopher D. Spicer, Albane Imbert, Francesco Saverio Tedesco, Andrea Serio
Published in: PLOS Biology, Issue 22, 2024, Page(s) e3002503, ISSN 1545-7885
Publisher: PLOS
DOI: 10.1371/journal.pbio.3002503

Ammonia transporter RhBG initiates downstream signaling and functional responses by activating NFκB

Author(s): Saurabh Mishra, Nicole Welch, Shashi Shekhar Singh, Khuraijam Dhanachandra Singh, Annette Bellar, Avinash Kumar, Lars N. Deutz, Maxmillian D. Hanlon, Sashi Kant, Sumitava Dastidar, Hailee Patel, Vandana Agrawal, Amy H. Attaway, Ryan Musich, George R. Stark, Francesco Saverio Tedesco, George A. Truskey, I. David Weiner, Sadashiva S. Karnik, Srinivasan Dasarathy
Published in: Proceedings of the National Academy of Sciences, Issue 121, 2024, ISSN 0027-8424
Publisher: National Academy of Sciences
DOI: 10.1073/pnas.2314760121

Generation of two genomic-integration-free DMD iPSC lines with mutations affecting all dystrophin isoforms and potentially amenable to exon-skipping

Author(s): Giulia Ferrari, Francesco Muntoni, Francesco Saverio Tedesco
Published in: Stem Cell Research, Issue 43, 2020, Page(s) 101688, ISSN 1873-5061
Publisher: Elsevier
DOI: 10.1016/j.scr.2019.101688

Investigating the role of dystrophin isoform deficiency in motor function in Duchenne muscular dystrophy

Author(s): Mary Chesshyre, Deborah Ridout, Yasumasa Hashimoto, Yoko Ookubo, Silvia Torelli, Kate Maresh, Valeria Ricotti, Lianne Abbott, Vandana Ayyar Gupta, Marion Main, Giulia Ferrari, Anna Kowala, Yung‐Yao Lin, Francesco Saverio Tedesco, Mariacristina Scoto, Giovanni Baranello, Adnan Manzur, Yoshitsugu Aoki, Francesco Muntoni
Published in: Journal of Cachexia, Sarcopenia and Muscle, Issue 13, 2023, Page(s) 1360-1372, ISSN 2190-5991
Publisher: Springer Verlag
DOI: 10.1002/jcsm.12914

Advanced models of human skeletal muscle differentiation, development and disease: Three-dimensional cultures, organoids and beyond

Author(s): Salma Jalal, Sumitava Dastidar, Francesco Saverio Tedesco
Published in: Current Opinion in Cell Biology, Issue 73, 2023, Page(s) 92-104, ISSN 0955-0674
Publisher: Elsevier BV
DOI: 10.1016/j.ceb.2021.06.004

Combined Notch and PDGF Signaling Enhances Migration and Expression of Stem Cell Markers while Inducing Perivascular Cell Features in Muscle Satellite Cells

Author(s): Mattia Francesco Maria Gerli, Louise Anne Moyle, Sara Benedetti, Giulia Ferrari, Ekin Ucuncu, Martina Ragazzi, Chrystalla Constantinou, Irene Louca, Hiroshi Sakai, Pierpaolo Ala, Paolo De Coppi, Shahragim Tajbakhsh, Giulio Cossu, Francesco Saverio Tedesco
Published in: Stem Cell Reports, Issue 12/3, 2019, Page(s) 461-473, ISSN 2213-6711
Publisher: Cell Press
DOI: 10.1016/j.stemcr.2019.01.007

Dystrophin deficiency affects human astrocyte properties and response to damage

Author(s): Jenny Lange, Olivia Gillham, Reem Alkharji, Simon Eaton, Giulia Ferrari, Monika Madej, Michael Flower, Francesco Saverio Tedesco, Francesco Muntoni, Patrizia Ferretti
Published in: Glia, Issue 70, 2023, Page(s) 466-490, ISSN 0894-1491
Publisher: John Wiley & Sons Inc.
DOI: 10.1002/glia.24116

Book chapters (1)

Pericytes in Muscular Dystrophies

Author(s): Louise Anne Moyle, Francesco Saverio Tedesco, Sara Benedetti
Published in: Pericyte Biology in Disease, Issue 1147, 2019, Page(s) 319-344, ISBN 978-3-030-16907-7
Publisher: Springer International Publishing
DOI: 10.1007/978-3-030-16908-4_15

Non-peer reviewed articles (2)

Challenging the “chromatin hypothesis” of cardiac laminopathies with LMNA mutant iPS cells

Author(s): Chiara Mozzetta, Francesco Saverio Tedesco
Published in: Journal of Cell Biology, Issue 218/9, 2019, Page(s) 2826-2828, ISSN 0021-9525
Publisher: Rockefeller University Press
DOI: 10.1083/jcb.201907166

Stem cell culture conditions and stability: a joint workshop of the PluriMes Consortium and Pluripotent Stem Cell Platform

Author(s): Glyn N Stacey, Peter W Andrews, Ivana Barbaric, Charlotta Boiers, Amit Chandra, Giulio Cossu, Lynn Csontos, Thomas JR Frith, Jason A Halliwell, Zoe Hewitt, Mark McCall, Harry D Moore, Malin Parmar, M Beatrice Panico, Venkat Pisupati, Valentin P Shichkin, Alison R Stacey, Francesco S Tedesco, Oliver Thompson, Ravenska Wagey
Published in: Regenerative Medicine, Issue 14/3, 2019, Page(s) 243-255, ISSN 1746-0751
Publisher: Future Medicine Ltd.
DOI: 10.2217/rme-2019-0001