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Screening identifies small molecules that enhance the maturation of human pluripotent stem cell-derived myotubes.

Selvaraj S, Mondragon-Gonzalez R, Xu B, Magli A, Kim H, Lainé J, Kiley J, Mckee H, Rinaldi F, Aho J, Tabti N, Shen W, Perlingeiro RC.

Elife. 2019 Nov 11;8. pii: e47970. doi: 10.7554/eLife.47970.


Gene Correction of LGMD2A Patient-Specific iPSCs for the Development of Targeted Autologous Cell Therapy.

Selvaraj S, Dhoke NR, Kiley J, Mateos-Aierdi AJ, Tungtur S, Mondragon-Gonzalez R, Killeen G, Oliveira VKP, López de Munain A, Perlingeiro RCR.

Mol Ther. 2019 Dec 4;27(12):2147-2157. doi: 10.1016/j.ymthe.2019.08.011. Epub 2019 Aug 28.


Pluripotent Stem Cell-Based Therapeutics for Muscular Dystrophies.

Selvaraj S, Kyba M, Perlingeiro RCR.

Trends Mol Med. 2019 Sep;25(9):803-816. doi: 10.1016/j.molmed.2019.07.004. Review.


Transplantation studies reveal internuclear transfer of toxic RNA in engrafted muscles of myotonic dystrophy 1 mice.

Mondragon-Gonzalez R, Azzag K, Selvaraj S, Yamamoto A, Perlingeiro RCR.

EBioMedicine. 2019 Sep;47:553-562. doi: 10.1016/j.ebiom.2019.08.031. Epub 2019 Aug 21.


Pax3 cooperates with Ldb1 to direct local chromosome architecture during myogenic lineage specification.

Magli A, Baik J, Pota P, Cordero CO, Kwak IY, Garry DJ, Love PE, Dynlacht BD, Perlingeiro RCR.

Nat Commun. 2019 May 24;10(1):2316. doi: 10.1038/s41467-019-10318-6.


Time-dependent Pax3-mediated chromatin remodeling and cooperation with Six4 and Tead2 specify the skeletal myogenic lineage in developing mesoderm.

Magli A, Baik J, Mills LJ, Kwak IY, Dillon BS, Mondragon Gonzalez R, Stafford DA, Swanson SA, Stewart R, Thomson JA, Garry DJ, Dynlacht BD, Perlingeiro RCR.

PLoS Biol. 2019 Feb 26;17(2):e3000153. doi: 10.1371/journal.pbio.3000153. eCollection 2019 Feb.


Pluripotent stem cell-derived myogenic progenitors remodel their molecular signature upon in vivo engraftment.

Incitti T, Magli A, Darabi R, Yuan C, Lin K, Arpke RW, Azzag K, Yamamoto A, Stewart R, Thomson JA, Kyba M, Perlingeiro RCR.

Proc Natl Acad Sci U S A. 2019 Mar 5;116(10):4346-4351. doi: 10.1073/pnas.1808303116. Epub 2019 Feb 13.


Serial transplantation reveals a critical role for endoglin in hematopoietic stem cell quiescence.

Borges L, Oliveira VKP, Baik J, Bendall SC, Perlingeiro RCR.

Blood. 2019 Feb 14;133(7):688-696. doi: 10.1182/blood-2018-09-874677. Epub 2018 Dec 28.


Nanotopography-responsive myotube alignment and orientation as a sensitive phenotypic biomarker for Duchenne Muscular Dystrophy.

Xu B, Magli A, Anugrah Y, Koester SJ, Perlingeiro RCR, Shen W.

Biomaterials. 2018 Nov;183:54-66. doi: 10.1016/j.biomaterials.2018.08.047. Epub 2018 Aug 21.


Skeletal Muscle Stem Cells from PSC-Derived Teratomas Have Functional Regenerative Capacity.

Chan SS, Arpke RW, Filareto A, Xie N, Pappas MP, Penaloza JS, Perlingeiro RCR, Kyba M.

Cell Stem Cell. 2018 Jul 5;23(1):74-85.e6. doi: 10.1016/j.stem.2018.06.010.


Recapitulating muscle disease phenotypes with myotonic dystrophy 1 induced pluripotent stem cells: a tool for disease modeling and drug discovery.

Mondragon-Gonzalez R, Perlingeiro RCR.

Dis Model Mech. 2018 Jul 18;11(7). pii: dmm034728. doi: 10.1242/dmm.034728.


The DUX4 homeodomains mediate inhibition of myogenesis and are functionally exchangeable with the Pax7 homeodomain.

Bosnakovski D, Toso EA, Hartweck LM, Magli A, Lee HA, Thompson ER, Dandapat A, Perlingeiro RCR, Kyba M.

J Cell Sci. 2017 Nov 1;130(21):3685-3697. doi: 10.1242/jcs.205427. Epub 2017 Sep 21.


Generation of skeletal myogenic progenitors from human pluripotent stem cells using non-viral delivery of minicircle DNA.

Kim J, Oliveira VKP, Yamamoto A, Perlingeiro RCR.

Stem Cell Res. 2017 Aug;23:87-94. doi: 10.1016/j.scr.2017.07.013. Epub 2017 Jul 12.


PAX7 Targets, CD54, Integrin α9β1, and SDC2, Allow Isolation of Human ESC/iPSC-Derived Myogenic Progenitors.

Magli A, Incitti T, Kiley J, Swanson SA, Darabi R, Rinaldi F, Selvaraj S, Yamamoto A, Tolar J, Yuan C, Stewart R, Thomson JA, Perlingeiro RCR.

Cell Rep. 2017 Jun 27;19(13):2867-2877. doi: 10.1016/j.celrep.2017.06.005.


Expansion and Purification Are Critical for the Therapeutic Application of Pluripotent Stem Cell-Derived Myogenic Progenitors.

Kim J, Magli A, Chan SSK, Oliveira VKP, Wu J, Darabi R, Kyba M, Perlingeiro RCR.

Stem Cell Reports. 2017 Jul 11;9(1):12-22. doi: 10.1016/j.stemcr.2017.04.022. Epub 2017 May 18.


Endoglin: a novel target for therapeutic intervention in acute leukemias revealed in xenograft mouse models.

Dourado KMC, Baik J, Oliveira VKP, Beltrame M, Yamamoto A, Theuer CP, Figueiredo CAV, Verneris MR, Perlingeiro RCR.

Blood. 2017 May 4;129(18):2526-2536. doi: 10.1182/blood-2017-01-763581. Epub 2017 Mar 28.


Endoglin integrates BMP and Wnt signalling to induce haematopoiesis through JDP2.

Baik J, Magli A, Tahara N, Swanson SA, Koyano-Nakagawa N, Borges L, Stewart R, Garry DJ, Kawakami Y, Thomson JA, Perlingeiro RC.

Nat Commun. 2016 Oct 7;7:13101. doi: 10.1038/ncomms13101.


Myogenic Progenitors from Mouse Pluripotent Stem Cells for Muscle Regeneration.

Magli A, Incitti T, Perlingeiro RC.

Methods Mol Biol. 2016;1460:191-208. doi: 10.1007/978-1-4939-3810-0_14.


Treatment with rGDF11 does not improve the dystrophic muscle pathology of mdx mice.

Rinaldi F, Zhang Y, Mondragon-Gonzalez R, Harvey J, Perlingeiro RCR.

Skelet Muscle. 2016 Jun 14;6:21. doi: 10.1186/s13395-016-0092-8. eCollection 2016.


Membrane-stabilizing copolymers confer marked protection to dystrophic skeletal muscle in vivo.

Houang EM, Haman KJ, Filareto A, Perlingeiro RC, Bates FS, Lowe DA, Metzger JM.

Mol Ther Methods Clin Dev. 2015 Nov 11;2:15042. doi: 10.1038/mtm.2015.42. eCollection 2015.


Pax3-induced expansion enables the genetic correction of dystrophic satellite cells.

Filareto A, Rinaldi F, Arpke RW, Darabi R, Belanto JJ, Toso EA, Miller AZ, Ervasti JM, McIvor RS, Kyba M, Perlingeiro RC.

Skelet Muscle. 2015 Oct 26;5:36. doi: 10.1186/s13395-015-0061-7. eCollection 2015.


Derivation of Skeletal Myogenic Precursors from Human Pluripotent Stem Cells Using Conditional Expression of PAX7.

Darabi R, Perlingeiro RC.

Methods Mol Biol. 2016;1357:423-39. doi: 10.1007/7651_2014_134.


Dominant lethal pathologies in male mice engineered to contain an X-linked DUX4 transgene.

Dandapat A, Bosnakovski D, Hartweck LM, Arpke RW, Baltgalvis KA, Vang D, Baik J, Darabi R, Perlingeiro RC, Hamra FK, Gupta K, Lowe DA, Kyba M.

Cell Rep. 2014 Sep 11;8(5):1484-96. doi: 10.1016/j.celrep.2014.07.056. Epub 2014 Aug 28.


Coaxing stem cells for skeletal muscle repair.

McCullagh KJ, Perlingeiro RC.

Adv Drug Deliv Rev. 2015 Apr;84:198-207. doi: 10.1016/j.addr.2014.07.007. Epub 2014 Jul 15. Review.


Pax3 and Tbx5 specify whether PDGFRα+ cells assume skeletal or cardiac muscle fate in differentiating embryonic stem cells.

Magli A, Schnettler E, Swanson SA, Borges L, Hoffman K, Stewart R, Thomson JA, Keirstead SA, Perlingeiro RC.

Stem Cells. 2014 Aug;32(8):2072-83. doi: 10.1002/stem.1713.


Stem cells for skeletal muscle regeneration: therapeutic potential and roadblocks.

Rinaldi F, Perlingeiro RC.

Transl Res. 2014 Apr;163(4):409-17. doi: 10.1016/j.trsl.2013.11.006. Epub 2013 Nov 14. Review.


Are we there yet? Navigating roadblocks to induced pluripotent stem cell therapy translation.

Parker SL, Perlingeiro RC.

Regen Med. 2013 Jul;8(4):389-91. doi: 10.2217/rme.13.37. No abstract available.


Expression levels of endoglin distinctively identify hematopoietic and endothelial progeny at different stages of yolk sac hematopoiesis.

Borges L, Iacovino M, Koyano-Nakagawa N, Baik J, Garry DJ, Kyba M, Perlingeiro RC.

Stem Cells. 2013 Sep;31(9):1893-901. doi: 10.1002/stem.1434.


A new immuno-, dystrophin-deficient model, the NSG-mdx(4Cv) mouse, provides evidence for functional improvement following allogeneic satellite cell transplantation.

Arpke RW, Darabi R, Mader TL, Zhang Y, Toyama A, Lonetree CL, Nash N, Lowe DA, Perlingeiro RC, Kyba M.

Stem Cells. 2013 Aug;31(8):1611-20. doi: 10.1002/stem.1402.


An ex vivo gene therapy approach to treat muscular dystrophy using inducible pluripotent stem cells.

Filareto A, Parker S, Darabi R, Borges L, Iacovino M, Schaaf T, Mayerhofer T, Chamberlain JS, Ervasti JM, McIvor RS, Kyba M, Perlingeiro RC.

Nat Commun. 2013;4:1549. doi: 10.1038/ncomms2550.


Functional dissection of Pax3 in paraxial mesoderm development and myogenesis.

Magli A, Schnettler E, Rinaldi F, Bremer P, Perlingeiro RC.

Stem Cells. 2013 Jan;31(1):59-70. doi: 10.1002/stem.1254.


Effect of endoglin overexpression during embryoid body development.

Baik J, Borges L, Magli A, Thatava T, Perlingeiro RC.

Exp Hematol. 2012 Oct;40(10):837-46. doi: 10.1016/j.exphem.2012.06.007. Epub 2012 Jun 19.


Etv2 is expressed in the yolk sac hematopoietic and endothelial progenitors and regulates Lmo2 gene expression.

Koyano-Nakagawa N, Kweon J, Iacovino M, Shi X, Rasmussen TL, Borges L, Zirbes KM, Li T, Perlingeiro RC, Kyba M, Garry DJ.

Stem Cells. 2012 Aug;30(8):1611-23. doi: 10.1002/stem.1131.


Human ES- and iPS-derived myogenic progenitors restore DYSTROPHIN and improve contractility upon transplantation in dystrophic mice.

Darabi R, Arpke RW, Irion S, Dimos JT, Grskovic M, Kyba M, Perlingeiro RC.

Cell Stem Cell. 2012 May 4;10(5):610-9. doi: 10.1016/j.stem.2012.02.015.


A critical role for endoglin in the emergence of blood during embryonic development.

Borges L, Iacovino M, Mayerhofer T, Koyano-Nakagawa N, Baik J, Garry DJ, Kyba M, Letarte M, Perlingeiro RC.

Blood. 2012 Jun 7;119(23):5417-28. doi: 10.1182/blood-2011-11-391896. Epub 2012 Apr 24.


Engraftment of ES-Derived Myogenic Progenitors in a Severe Mouse Model of Muscular Dystrophy.

Filareto A, Darabi R, Perlingeiro RC.

J Stem Cell Res Ther. 2012 Jan 6;10(1). pii: S10-001.


Modulation of TGF-β signaling by endoglin in murine hemangioblast development and primitive hematopoiesis.

Zhang L, Magli A, Catanese J, Xu Z, Kyba M, Perlingeiro RC.

Blood. 2011 Jul 7;118(1):88-97. doi: 10.1182/blood-2010-12-325019. Epub 2011 May 20.


Functional myogenic engraftment from mouse iPS cells.

Darabi R, Pan W, Bosnakovski D, Baik J, Kyba M, Perlingeiro RC.

Stem Cell Rev Rep. 2011 Nov;7(4):948-57. doi: 10.1007/s12015-011-9258-2.


Assessment of the myogenic stem cell compartment following transplantation of Pax3/Pax7-induced embryonic stem cell-derived progenitors.

Darabi R, Santos FN, Filareto A, Pan W, Koene R, Rudnicki MA, Kyba M, Perlingeiro RC.

Stem Cells. 2011 May;29(5):777-90. doi: 10.1002/stem.625.


Clonal analysis reveals a common progenitor for endothelial, myeloid, and lymphoid precursors in umbilical cord blood.

Ramos AL, Darabi R, Akbarloo N, Borges L, Catanese J, Dineen SP, Brekken RA, Perlingeiro RC.

Circ Res. 2010 Dec 10;107(12):1460-9. doi: 10.1161/CIRCRESAHA.110.223669. Epub 2010 Oct 14.


Engraftment of embryonic stem cell-derived myogenic progenitors in a dominant model of muscular dystrophy.

Darabi R, Baik J, Clee M, Kyba M, Tupler R, Perlingeiro RC.

Exp Neurol. 2009 Nov;220(1):212-6. doi: 10.1016/j.expneurol.2009.08.002. Epub 2009 Aug 13.


Engraftment of mesenchymal stem cells into dystrophin-deficient mice is not accompanied by functional recovery.

Gang EJ, Darabi R, Bosnakovski D, Xu Z, Kamm KE, Kyba M, Perlingeiro RC.

Exp Cell Res. 2009 Sep 10;315(15):2624-36. doi: 10.1016/j.yexcr.2009.05.009. Epub 2009 May 19.


Mesodermal patterning activity of SCL.

Ismailoglu I, Yeamans G, Daley GQ, Perlingeiro RC, Kyba M.

Exp Hematol. 2008 Dec;36(12):1593-603. doi: 10.1016/j.exphem.2008.07.005. Epub 2008 Sep 21.


Prospective isolation of skeletal muscle stem cells with a Pax7 reporter.

Bosnakovski D, Xu Z, Li W, Thet S, Cleaver O, Perlingeiro RC, Kyba M.

Stem Cells. 2008 Dec;26(12):3194-204. doi: 10.1634/stemcells.2007-1017. Epub 2008 Sep 18.


The therapeutic potential of embryonic and adult stem cells for skeletal muscle regeneration.

Darabi R, Santos FN, Perlingeiro RC.

Stem Cell Rev. 2008 Sep;4(3):217-25. doi: 10.1007/s12015-008-9023-3. Epub 2008 Jul 8. Review.


Lineage-specific reprogramming as a strategy for cell therapy.

Darabi R, Perlingeiro RC.

Cell Cycle. 2008 Jun 15;7(12):1732-7. Epub 2008 Jun 11.


Pax3 activation promotes the differentiation of mesenchymal stem cells toward the myogenic lineage.

Gang EJ, Bosnakovski D, Simsek T, To K, Perlingeiro RC.

Exp Cell Res. 2008 May 1;314(8):1721-33. doi: 10.1016/j.yexcr.2008.02.016. Epub 2008 Mar 5.


Functional skeletal muscle regeneration from differentiating embryonic stem cells.

Darabi R, Gehlbach K, Bachoo RM, Kamath S, Osawa M, Kamm KE, Kyba M, Perlingeiro RC.

Nat Med. 2008 Feb;14(2):134-43. doi: 10.1038/nm1705. Epub 2008 Jan 20.


Endoglin is required for hemangioblast and early hematopoietic development.

Perlingeiro RC.

Development. 2007 Aug;134(16):3041-8. Epub 2007 Jul 18.

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