{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,31]],"date-time":"2026-10-31T03:56:33Z","timestamp":1793418993751,"version":"4.5.0"},"reference-count":137,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[2024,11,5]],"date-time":"2024-11-05T00:00:00Z","timestamp":1730764800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["20H03885"],"award-info":[{"award-number":["20H03885"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["21H03142"],"award-info":[{"award-number":["21H03142"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["21\u00a0K19589"],"award-info":[{"award-number":["21\u00a0K19589"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Rising Star Award from American Society for Bone and Mineral Research"},{"DOI":"10.13039\/100009619","name":"Japan Agency for Medical Research and Development","doi-asserted-by":"publisher","award":["JP21bm0704071"],"award-info":[{"award-number":["JP21bm0704071"]}],"id":[{"id":"10.13039\/100009619","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100002241","name":"Japan Science and Technology Agency","doi-asserted-by":"publisher","award":["JPMJFR225N"],"award-info":[{"award-number":["JPMJFR225N"]}],"id":[{"id":"10.13039\/501100002241","id-type":"DOI","asserted-by":"publisher"}]},{"name":"JST ERATO program","award":["JPMJER2401"],"award-info":[{"award-number":["JPMJER2401"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,12,31]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Human skeletal elements are formed from distinct origins at distinct positions of the embryo. For example, the neural crest produces the facial bones, the paraxial mesoderm produces the axial skeleton, and the lateral plate mesoderm produces the appendicular skeleton. During skeletal development, different combinations of signaling pathways are coordinated from distinct origins during the sequential developmental stages. Models for human skeletal development have been established using human pluripotent stem cells (hPSCs) and by exploiting our understanding of skeletal development. Stepwise protocols for generating skeletal cells from different origins have been designed to mimic developmental trails. Recently, organoid methods have allowed the multicellular organization of skeletal cell types to recapitulate complicated skeletal development and metabolism. Similarly, several genetic diseases of the skeleton have been modeled using patient-derived induced pluripotent stem cells and genome-editing technologies. Model-based drug screening is a powerful tool for identifying drug candidates. This review briefly summarizes our current understanding of the embryonic development of skeletal tissues and introduces the current state-of-the-art hPSC methods for recapitulating skeletal development, metabolism, and diseases. We also discuss the current limitations and future perspectives for applications of the hPSC-based modeling system in precision medicine in this research field.<\/jats:p>","DOI":"10.1093\/jbmr\/zjae178","type":"journal-article","created":{"date-parts":[[2024,11,5]],"date-time":"2024-11-05T09:25:57Z","timestamp":1730798757000},"page":"5-19","source":"Crossref","is-referenced-by-count":12,"title":["Modeling of skeletal development and diseases using human pluripotent stem cells"],"prefix":"10.1093","volume":"40","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2245-6853","authenticated-orcid":false,"given":"Hironori","family":"Hojo","sequence":"first","affiliation":[{"name":"Division of Clinical Biotechnology , Center for Disease Biology and Integrative Medicine, Graduate School of Medicine, , Tokyo 113-8655,","place":["Japan"]},{"name":"The University of Tokyo , Center for Disease Biology and Integrative Medicine, Graduate School of Medicine, , Tokyo 113-8655,","place":["Japan"]},{"name":"Department of Bioengineering , Graduate School of Engineering, , Tokyo 113-8655,","place":["Japan"]},{"name":"The University of Tokyo , Graduate School of Engineering, , Tokyo 113-8655,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shoichiro","family":"Tani","sequence":"additional","affiliation":[{"name":"Children\u2019s Medical Center Research Institute, University of Texas Southwestern Medical Center , Dallas, TX 75390,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shinsuke","family":"Ohba","sequence":"additional","affiliation":[{"name":"Department of Tissue and Developmental Biology , Graduate School of Dentistry, , Osaka 565-0871,","place":["Japan"]},{"name":"Osaka University , Graduate School of Dentistry, , Osaka 565-0871,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2024,11,5]]},"reference":[{"key":"2025010523412045100_ref1","volume-title":"Vertebrate Skeletal Development","author":"Olsen","year":"2019","edition":"First"},{"issue":"3","key":"2025010523412045100_ref2","doi-asserted-by":"publisher","first-page":"414","DOI":"10.1002\/dvdy.278","article-title":"Making and shaping endochondral and intramembranous bones","volume":"250","author":"Galea","year":"2021","journal-title":"Developmental dynamics : an official publication of the American Association of Anatomists"},{"key":"2025010523412045100_ref3","doi-asserted-by":"publisher","first-page":"19","DOI":"10.1016\/B978-0-323-85545-7.00010-7","volume-title":"iPSC Derived Progenitors. 13","author":"Srinivasan","year":"2022"},{"key":"2025010523412045100_ref4","doi-asserted-by":"publisher","first-page":"1056114","DOI":"10.3389\/fgene.2022.1056114","article-title":"Mesoangioblasts at 20: from the embryonic aorta to the patient bed","volume":"13","author":"Cossu","year":"2022","journal-title":"Front Genet"},{"issue":"6","key":"2025010523412045100_ref5","doi-asserted-by":"publisher","first-page":"815","DOI":"10.1007\/s11914-023-00827-1","article-title":"The origin and fate of chondrocytes: cell plasticity in physiological setting","volume":"21","author":"Chagin","year":"2023","journal-title":"Current Osteoporosis Reports"},{"key":"2025010523412045100_ref6","article-title":"MT1-MMP-dependent, apoptotic remodeling of unmineralized cartilage: a critical process in skeletal growth","volume":"163","author":"Holmbeck","journal-title":"J Cell Biol"},{"issue":"7725","key":"2025010523412045100_ref7","doi-asserted-by":"publisher","first-page":"133","DOI":"10.1038\/s41586-018-0554-8","article-title":"Discovery of a periosteal stem cell mediating intramembranous bone formation","volume":"562","author":"Debnath","year":"2018","journal-title":"Nature"},{"issue":"1","key":"2025010523412045100_ref8","doi-asserted-by":"publisher","first-page":"207","DOI":"10.1021\/acsbiomaterials.0c00878","article-title":"Comparative craniofacial bone regeneration capacities of mesenchymal stem cells derived from human neural crest stem cells and bone marrow","volume":"7","author":"Srinivasan","year":"2021","journal-title":"ACS Biomater Sci Eng"},{"issue":"6255","key":"2025010523412045100_ref9","doi-asserted-by":"publisher","first-page":"273","DOI":"10.1038\/343273a0","article-title":"Origin of cells giving rise to mesoderm and endoderm in chick embryo","volume":"343","author":"Stern","year":"1990","journal-title":"Nature"},{"issue":"4","key":"2025010523412045100_ref10","doi-asserted-by":"publisher","first-page":"661","DOI":"10.1016\/j.cell.2008.02.008","article-title":"Differentiation of embryonic stem cells to clinically relevant populations: lessons from embryonic development","volume":"132","author":"Murry","year":"2008","journal-title":"Cell"},{"issue":"1-2","key":"2025010523412045100_ref11","doi-asserted-by":"publisher","first-page":"3","DOI":"10.1016\/S0925-4773(97)00123-8","article-title":"Mouse gastrulation: the formation of a mammalian body plan","volume":"68","author":"Tam","year":"1997","journal-title":"Mech Dev"},{"issue":"8","key":"2025010523412045100_ref12","doi-asserted-by":"publisher","first-page":"1166","DOI":"10.1038\/s12276-020-0482-1","article-title":"Understanding paraxial mesoderm development and sclerotome specification for skeletal repair","volume":"52","author":"Tani","year":"2020","journal-title":"Exp Mol Med"},{"issue":"5","key":"2025010523412045100_ref13","doi-asserted-by":"publisher","first-page":"547","DOI":"10.1002\/dvdy.24392","article-title":"Notochord morphogenesis in mice: current understanding & open questions","volume":"245","author":"Balmer","year":"2016","journal-title":"Dev Dyn"},{"issue":"3","key":"2025010523412045100_ref14","doi-asserted-by":"publisher","first-page":"313","DOI":"10.1016\/j.devcel.2006.07.005","article-title":"The nodal precursor acting via activin receptors induces mesoderm by maintaining a source of its convertases and BMP4","volume":"11","author":"Ben-Haim","year":"2006","journal-title":"Dev Cell"},{"key":"2025010523412045100_ref15","doi-asserted-by":"publisher","first-page":"73","DOI":"10.1016\/j.semcdb.2014.03.028","article-title":"Dose-dependent Nodal\/Smad signals pattern the early mouse embryo","volume":"32","author":"Robertson","year":"2014","journal-title":"Semin Cell Dev Biol"},{"issue":"17","key":"2025010523412045100_ref16","doi-asserted-by":"publisher","first-page":"2105","DOI":"10.1101\/gad.9.17.2105","article-title":"Bone morphogenetic protein-4 is required for mesoderm formation and patterning in the mouse","volume":"9","author":"Winnier","year":"1995","journal-title":"Genes Dev"},{"issue":"2","key":"2025010523412045100_ref17","doi-asserted-by":"publisher","first-page":"174","DOI":"10.1101\/gad.8.2.174","article-title":"Wnt-3a regulates somite and tailbud formation in the mouse embryo","volume":"8","author":"Takada","year":"1994","journal-title":"Genes Dev"},{"issue":"6","key":"2025010523412045100_ref18","doi-asserted-by":"publisher","first-page":"1573","DOI":"10.1016\/j.celrep.2017.01.040","article-title":"In vivo human somitogenesis guides somite development from hPSCs","volume":"18","author":"Xi","year":"2017","journal-title":"Cell Rep"},{"issue":"3","key":"2025010523412045100_ref19","doi-asserted-by":"publisher","first-page":"383","DOI":"10.1038\/ng0794-383","article-title":"An ancient family of embryonically expressed mouse genes sharing a conserved protein motif with the T locus","volume":"7","author":"Bollag","year":"1994","journal-title":"Nat Genet"},{"issue":"8","key":"2025010523412045100_ref20","doi-asserted-by":"publisher","first-page":"784","DOI":"10.1038\/sj.embor.7401030","article-title":"Expression of Msgn1 in the presomitic mesoderm is controlled by synergism of WNT signalling and Tbx6","volume":"8","author":"Wittler","year":"2007","journal-title":"EMBO Rep"},{"issue":"2","key":"2025010523412045100_ref21","doi-asserted-by":"publisher","first-page":"172","DOI":"10.1006\/dbio.1998.8895","article-title":"Somitogenesis controlled by noggin","volume":"202","author":"Tonegawa","year":"1998","journal-title":"Dev Biol"},{"issue":"5","key":"2025010523412045100_ref22","doi-asserted-by":"publisher","first-page":"370","DOI":"10.1038\/nrg2320","article-title":"Segmental patterning of the vertebrate embryonic axis","volume":"9","author":"Dequ\u00e9ant","year":"2008","journal-title":"Nat Rev Genet"},{"issue":"2","key":"2025010523412045100_ref23","doi-asserted-by":"publisher","first-page":"a000869","DOI":"10.1101\/cshperspect.a000869","article-title":"Signaling gradients during paraxial mesoderm development","volume":"2","author":"Aulehla","year":"2010","journal-title":"Cold Spring Harb Perspect Biol"},{"issue":"6599","key":"2025010523412045100_ref24","doi-asserted-by":"publisher","first-page":"407","DOI":"10.1038\/383407a0","article-title":"Cyclopia and defective axial patterning in mice lacking sonic hedgehog gene function","volume":"383","author":"Chiang","year":"1996","journal-title":"Nature"},{"issue":"15","key":"2025010523412045100_ref25","doi-asserted-by":"publisher","first-page":"1990","DOI":"10.1101\/gad.1008002","article-title":"Shh establishes an Nkx3. 2\/Sox9 autoregulatory loop that is maintained by BMP signals to induce somitic chondrogenesis","volume":"16","author":"Zeng","year":"2002","journal-title":"Genes Dev"},{"issue":"5","key":"2025010523412045100_ref26","doi-asserted-by":"publisher","first-page":"1005","DOI":"10.1242\/dev.051938","article-title":"Cooperative activity of noggin and gremlin 1 in axial skeleton development","volume":"138","author":"Stafford","year":"2011","journal-title":"Development"},{"key":"2025010523412045100_ref27","doi-asserted-by":"publisher","first-page":"39","DOI":"10.1016\/j.ceb.2019.07.008","article-title":"SOX9 in cartilage development and disease","volume":"61","author":"Lefebvre","year":"2019","journal-title":"Curr Opin Cell Biol"},{"issue":"12","key":"2025010523412045100_ref28","doi-asserted-by":"publisher","first-page":"774","DOI":"10.1016\/j.tig.2016.10.001","article-title":"An emerging regulatory landscape for skeletal development","volume":"32","author":"Hojo","year":"2016","journal-title":"Trends in genetics : TIG"},{"issue":"5","key":"2025010523412045100_ref29","doi-asserted-by":"crossref","first-page":"817","DOI":"10.1242\/dev.105536","article-title":"A pathway to bone: signaling molecules and transcription factors involved in chondrocyte development and maturation","volume":"142","author":"Kozhemyakina","year":"2015","journal-title":"Development"},{"key":"2025010523412045100_ref30","doi-asserted-by":"publisher","first-page":"127","DOI":"10.1016\/B978-0-12-401730-6.00008-9","volume-title":"Neural Crest Cells","author":"Schilling","year":"2014"},{"issue":"18","key":"2025010523412045100_ref31","doi-asserted-by":"publisher","first-page":"4129","DOI":"10.1242\/dev.126.18.4129","article-title":"Coelom formation: binary decision of the lateral plate mesoderm is controlled by the ectoderm","volume":"126","author":"Funayama","year":"1999","journal-title":"Development"},{"key":"2025010523412045100_ref32","doi-asserted-by":"publisher","first-page":"689","DOI":"10.1242\/dev.01617","article-title":"Lineage analysis of the avian dermomyotome sheet reveals the existence of single cells with both dermal and muscle progenitor fates","volume-title":"Development","author":"Ben-Yair","year":"2005"},{"issue":"12","key":"2025010523412045100_ref33","doi-asserted-by":"publisher","DOI":"10.1242\/dev.175059","article-title":"The lateral plate mesoderm","volume":"147","author":"Prummel","year":"2020","journal-title":"Development"},{"key":"2025010523412045100_ref34","doi-asserted-by":"publisher","first-page":"1135784","DOI":"10.3389\/fcell.2023.1135784","article-title":"How might we build limbs in vitro informed by the modular aspects and tissue-dependency in limb development?","volume":"11","author":"Tsutsumi","year":"2023","journal-title":"Front Cell Dev Biol"},{"issue":"5","key":"2025010523412045100_ref35","doi-asserted-by":"publisher","first-page":"879","DOI":"10.1016\/j.celrep.2015.06.068","article-title":"RA acts in a coherent feed-forward mechanism with Tbx5 to control limb bud induction and initiation","volume":"12","author":"Nishimoto","year":"2015","journal-title":"Cell Rep"},{"issue":"19","key":"2025010523412045100_ref36","doi-asserted-by":"publisher","DOI":"10.1242\/dev.199580","article-title":"Tbx4 function during hindlimb development reveals a mechanism that explains the origins of proximal limb defects","volume":"148","author":"Duboc","year":"2021","journal-title":"Development"},{"issue":"3","key":"2025010523412045100_ref37","doi-asserted-by":"publisher","first-page":"260","DOI":"10.1038\/10320","article-title":"Regulation of anterior\/posterior patterning of the axial skeleton by growth\/differentiation factor 11","volume":"22","author":"McPherron","year":"1999","journal-title":"Nat Genet"},{"issue":"7193","key":"2025010523412045100_ref38","doi-asserted-by":"publisher","first-page":"401","DOI":"10.1038\/nature06876","article-title":"Genetic evidence that FGFs have an instructive role in limb proximal\u2013distal patterning","volume":"453","author":"Mariani","year":"2008","journal-title":"Nature"},{"issue":"6753","key":"2025010523412045100_ref39","doi-asserted-by":"publisher","first-page":"598","DOI":"10.1038\/44157","article-title":"Signal relay by BMP antagonism controls the SHH\/FGF4 feedback loop in vertebrate limb buds","volume":"401","author":"Z\u00fa\u00f1iga","year":"1999","journal-title":"Nature"},{"key":"2025010523412045100_ref40","doi-asserted-by":"publisher","first-page":"3401","DOI":"10.1242\/dev.01251","article-title":"Gremlin-mediated BMP antagonism induces the epithelial-mesenchymal feedback signaling controlling metanephric kidney and limb organogenesis","volume-title":"Development","author":"Michos","year":"2004"},{"issue":"7204","key":"2025010523412045100_ref41","doi-asserted-by":"publisher","first-page":"638","DOI":"10.1038\/nature07085","article-title":"An Fgf\/gremlin inhibitory feedback loop triggers termination of limb bud outgrowth","volume":"454","author":"Verheyden","year":"2008","journal-title":"Nature"},{"key":"2025010523412045100_ref42","doi-asserted-by":"publisher","first-page":"119","DOI":"10.1016\/j.semcdb.2014.04.004","article-title":"To BMP or not to BMP during vertebrate limb bud development","volume":"32","author":"Pignatti","year":"2014","journal-title":"Semin Cell Dev Biol"},{"issue":"2","key":"2025010523412045100_ref43","doi-asserted-by":"publisher","first-page":"77","DOI":"10.1002\/gene.10092","article-title":"Expression of Cre recombinase in the developing mouse limb bud driven by a Prxl enhancer","volume":"33","author":"Logan","year":"2002","journal-title":"Genesis (New York, NY: 2000)"},{"issue":"41","key":"2025010523412045100_ref44","doi-asserted-by":"publisher","first-page":"14665","DOI":"10.1073\/pnas.0504750102","article-title":"Osteo-chondroprogenitor cells are derived from Sox9 expressing precursors","volume":"102","author":"Akiyama","year":"2005","journal-title":"Proc Natl Acad Sci"},{"issue":"7","key":"2025010523412045100_ref45","doi-asserted-by":"publisher","first-page":"565","DOI":"10.1016\/j.devcel.2023.02.013","article-title":"A single-cell census of mouse limb development identifies complex spatiotemporal dynamics of skeleton formation","volume":"58","author":"Markman","year":"2023","journal-title":"Dev Cell"},{"issue":"7","key":"2025010523412045100_ref46","doi-asserted-by":"publisher","first-page":"742","DOI":"10.1038\/s41422-021-00467-z","article-title":"Dissecting human embryonic skeletal stem cell ontogeny by single-cell transcriptomic and functional analyses","volume":"31","author":"He","year":"2021","journal-title":"Cell Res"},{"issue":"10","key":"2025010523412045100_ref47","doi-asserted-by":"publisher","first-page":"616","DOI":"10.1038\/s41583-021-00503-2","article-title":"Riding the crest to get a head: neural crest evolution in vertebrates","volume":"22","author":"Martik","year":"2021","journal-title":"Nat Rev Neurosci"},{"issue":"1","key":"2025010523412045100_ref48","doi-asserted-by":"publisher","first-page":"e23276","DOI":"10.1002\/dvg.23276","article-title":"Specification and formation of the neural crest: perspectives on lineage segregation","volume":"57","author":"Prasad","year":"2019","journal-title":"Genesis"},{"key":"2025010523412045100_ref49","doi-asserted-by":"publisher","first-page":"17","DOI":"10.1016\/j.semcdb.2021.11.024","article-title":"Developmental principles informing human pluripotent stem cell differentiation to cartilage and bone","volume":"127","author":"Humphreys","year":"2022","journal-title":"Semin Cell Dev Biol"},{"issue":"2","key":"2025010523412045100_ref50","doi-asserted-by":"publisher","first-page":"242","DOI":"10.1242\/dev.105445","article-title":"Establishing neural crest identity: a gene regulatory recipe","volume":"142","author":"Sim\u00f5es-Costa","year":"2015","journal-title":"Development"},{"issue":"3","key":"2025010523412045100_ref51","doi-asserted-by":"publisher","first-page":"716","DOI":"10.1002\/dvdy.21881","article-title":"Comprehensive spatiotemporal analysis of early chick neural crest network genes","volume":"238","author":"Khudyakov","year":"2009","journal-title":"Dev Dyn"},{"issue":"14","key":"2025010523412045100_ref52","doi-asserted-by":"publisher","first-page":"5325","DOI":"10.1073\/pnas.85.14.5325","article-title":"Clone-forming ability and differentiation potential of migratory neural crest cells","volume":"85","author":"Baroffio","year":"1988","journal-title":"Proc Natl Acad Sci"},{"issue":"5","key":"2025010523412045100_ref53","doi-asserted-by":"publisher","first-page":"755","DOI":"10.1016\/S0092-8674(00)80258-5","article-title":"Targeted disruption of Cbfa1 results in a complete lack of bone formation owing to maturational arrest of osteoblasts","volume":"89","author":"Komori","year":"1997","journal-title":"Cell"},{"issue":"1","key":"2025010523412045100_ref54","doi-asserted-by":"publisher","first-page":"17","DOI":"10.1016\/S0092-8674(01)00622-5","article-title":"The novel zinc finger-containing transcription factor osterix is required for osteoblast differentiation and bone formation","volume":"108","author":"Nakashima","year":"2002","journal-title":"Cell"},{"issue":"2","key":"2025010523412045100_ref55","doi-asserted-by":"publisher","first-page":"138","DOI":"10.1016\/j.yexcr.2014.01.019","article-title":"Neural crest cell signaling pathways critical to cranial bone development and pathology","volume":"325","author":"Mishina","year":"2014","journal-title":"Exp Cell Res"},{"issue":"665","key":"2025010523412045100_ref56","doi-asserted-by":"publisher","DOI":"10.1126\/scisignal.aaz9368","article-title":"Augmented BMP signaling commits cranial neural crest cells to a chondrogenic fate by suppressing autophagic \u03b2-catenin degradation","volume":"14","author":"Yang","year":"2021","journal-title":"Sci Signal"},{"issue":"11","key":"2025010523412045100_ref57","doi-asserted-by":"publisher","first-page":"e14033","DOI":"10.1371\/journal.pone.0014033","article-title":"Activation of FGF Signaling mediates proliferative and osteogenic differences between neural crest derived frontal and mesoderm parietal derived bone","volume":"5","author":"Li","year":"2010","journal-title":"PLoS One"},{"issue":"47","key":"2025010523412045100_ref58","doi-asserted-by":"publisher","first-page":"40575","DOI":"10.1074\/jbc.M111.274910","article-title":"Fibroblast growth factor 2 stimulation of osteoblast differentiation and bone formation is mediated by modulation of the Wnt Signaling pathway*","volume":"286","author":"Fei","year":"2011","journal-title":"J Biol Chem"},{"issue":"8","key":"2025010523412045100_ref59","doi-asserted-by":"publisher","first-page":"1253","DOI":"10.1242\/dev.128.8.1253","article-title":"Inactivation of the \u03b2-catenin gene by Wnt1-Cre-mediated deletion results in dramatic brain malformation and failure of craniofacial development","volume":"128","author":"Brault","year":"2001","journal-title":"Development"},{"issue":"1","key":"2025010523412045100_ref60","doi-asserted-by":"crossref","first-page":"455","DOI":"10.1038\/srep00455","article-title":"Human chondrogenic paraxial mesoderm, directed specification and prospective isolation from pluripotent stem cells","volume":"2","author":"Umeda","year":"2012","journal-title":"Sci Rep"},{"issue":"1","key":"2025010523412045100_ref61","doi-asserted-by":"crossref","first-page":"66","DOI":"10.1186\/s13287-020-01597-8","article-title":"Prospective isolation of chondroprogenitors from human iPSCs based on cell surface markers identified using a CRISPR-Cas9-generated reporter","volume":"11","author":"Dicks","year":"2020","journal-title":"Stem Cell Res Ther"},{"issue":"6","key":"2025010523412045100_ref62","doi-asserted-by":"publisher","first-page":"638","DOI":"10.1038\/nbt.3210","article-title":"Generation of articular chondrocytes from human pluripotent stem cells","volume":"33","author":"Craft","year":"2015","journal-title":"Nat Biotechnol"},{"issue":"2","key":"2025010523412045100_ref63","doi-asserted-by":"publisher","first-page":"451","DOI":"10.1016\/j.cell.2016.06.011","article-title":"Mapping the pairwise choices leading from pluripotency to human bone, heart, and other mesoderm cell types","volume":"166","author":"Loh","year":"2016","journal-title":"Cell"},{"issue":"1","key":"2025010523412045100_ref64","doi-asserted-by":"publisher","first-page":"362","DOI":"10.1038\/s41467-020-20598-y","article-title":"Single cell transcriptomic analysis of human pluripotent stem cell chondrogenesis","volume":"12","author":"Wu","year":"2021","journal-title":"Nat Commun"},{"issue":"1","key":"2025010523412045100_ref65","doi-asserted-by":"publisher","first-page":"e0280024","DOI":"10.1371\/journal.pone.0280024","article-title":"Directed differentiation of hPSCs through a simplified lateral plate mesoderm protocol for generation of articular cartilage progenitors","volume":"18","author":"Smith","year":"2023","journal-title":"PLoS One"},{"issue":"4","key":"2025010523412045100_ref66","doi-asserted-by":"publisher","first-page":"712","DOI":"10.1016\/j.stemcr.2015.02.012","article-title":"Long-term expandable SOX9+ Chondrogenic Ectomesenchymal cells from human pluripotent stem cells","volume":"4","author":"Umeda","year":"2015","journal-title":"Stem cell reports"},{"issue":"1","key":"2025010523412045100_ref67","doi-asserted-by":"publisher","first-page":"69","DOI":"10.1038\/s41536-022-00265-0","article-title":"Rapid induction and long-term self-renewal of neural crest-derived ectodermal chondrogenic cells from hPSCs","volume":"7","author":"Shen","year":"2022","journal-title":"NPJ Regen Med"},{"issue":"9","key":"2025010523412045100_ref68","doi-asserted-by":"publisher","first-page":"1107","DOI":"10.1002\/stem.3206","article-title":"Lineage-specific differentiation of osteogenic progenitors from pluripotent stem cells reveals the FGF1-RUNX2 association in neural crest-derived osteoprogenitors","volume":"38","author":"Kidwai","year":"2020","journal-title":"Stem cells (Dayton, Ohio)"},{"issue":"4","key":"2025010523412045100_ref69","doi-asserted-by":"publisher","first-page":"112276","DOI":"10.1016\/j.celrep.2023.112276","article-title":"Stem cell-based modeling and single-cell multiomics reveal gene-regulatory mechanisms underlying human skeletal development","volume":"42","author":"Tani","year":"2023","journal-title":"Cell Rep"},{"issue":"19","key":"2025010523412045100_ref70","doi-asserted-by":"publisher","first-page":"e2211510120","DOI":"10.1073\/pnas.2211510120","article-title":"Modeling human skeletal development using human pluripotent stem cells","volume":"120","author":"Lamand\u00e9","year":"2023","journal-title":"Proc Natl Acad Sci USA"},{"issue":"45","key":"2025010523412045100_ref71","doi-asserted-by":"publisher","first-page":"16806","DOI":"10.1073\/pnas.0603916103","article-title":"Wnt and TGF-\u03b2 signaling are required for the induction of an in vitro model of primitive streak formation using embryonic stem cells","volume":"103","author":"Gadue","year":"2006","journal-title":"Proc Natl Acad Sci"},{"issue":"12","key":"2025010523412045100_ref72","doi-asserted-by":"publisher","first-page":"1534","DOI":"10.1038\/nbt1163","article-title":"Efficient differentiation of human embryonic stem cells to definitive endoderm","volume":"23","author":"D'Amour","year":"2005","journal-title":"Nat Biotechnol"},{"issue":"7801","key":"2025010523412045100_ref73","doi-asserted-by":"publisher","first-page":"124","DOI":"10.1038\/s41586-020-2144-9","article-title":"Recapitulating the human segmentation clock with pluripotent stem cells","volume":"580","author":"Matsuda","year":"2020","journal-title":"Nature"},{"issue":"9","key":"2025010523412045100_ref74","doi-asserted-by":"publisher","first-page":"2247","DOI":"10.1016\/j.celrep.2019.07.090","article-title":"An in vitro human segmentation clock model derived from embryonic stem cells","volume":"28","author":"Chu","year":"2019","journal-title":"Cell Rep"},{"issue":"2","key":"2025010523412045100_ref75","doi-asserted-by":"publisher","first-page":"134","DOI":"10.1038\/nm1705","article-title":"Functional skeletal muscle regeneration from differentiating embryonic stem cells","volume":"14","author":"Darabi","year":"2008","journal-title":"Nat Med"},{"key":"2025010523412045100_ref76","doi-asserted-by":"publisher","first-page":"17","DOI":"10.1016\/j.semcdb.2021.11.024","article-title":"Developmental principles informing human pluripotent stem cell differentiation to cartilage and bone","volume":"127","author":"Humphreys","year":"2022","journal-title":"Semin Cell Dev Biol"},{"issue":"3","key":"2025010523412045100_ref77","doi-asserted-by":"publisher","first-page":"397","DOI":"10.1002\/jbmr.4524","article-title":"Differentiation of induced pluripotent stem cells into chondrocytes: methods and applications for disease Modeling and drug discovery","volume":"37","author":"De Kinderen","year":"2022","journal-title":"Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research"},{"issue":"6","key":"2025010523412045100_ref78","doi-asserted-by":"publisher","DOI":"10.1242\/dev.157339","article-title":"Recapitulating early development of mouse musculoskeletal precursors of the paraxial mesoderm in vitro","volume":"145","author":"Chal","year":"2018","journal-title":"Development"},{"issue":"16","key":"2025010523412045100_ref79","doi-asserted-by":"publisher","DOI":"10.1242\/dev.165431","article-title":"Modeling human somite development and fibrodysplasia ossificans progressiva with induced pluripotent stem cells","volume":"145","author":"Nakajima","year":"2018","journal-title":"Development"},{"issue":"9","key":"2025010523412045100_ref80","doi-asserted-by":"publisher","first-page":"962","DOI":"10.1038\/nbt.3297","article-title":"Differentiation of pluripotent stem cells to muscle fiber to model Duchenne muscular dystrophy","volume":"33","author":"Chal","year":"2015","journal-title":"Nat Biotechnol"},{"issue":"10","key":"2025010523412045100_ref81","doi-asserted-by":"publisher","first-page":"1438","DOI":"10.1101\/gad.12.10.1438","article-title":"Noggin-mediated antagonism of BMP signaling is required for growth and patterning of the neural tube and somite","volume":"12","author":"McMahon","year":"1998","journal-title":"Genes Dev"},{"issue":"2","key":"2025010523412045100_ref82","doi-asserted-by":"publisher","first-page":"152","DOI":"10.1016\/j.ydbio.2008.08.024","article-title":"A gradient of Shh establishes mutually repressing somitic cell fates induced by Nkx3. 2 and Pax3","volume":"323","author":"Cairns","year":"2008","journal-title":"Dev Biol"},{"key":"2025010523412045100_ref83","doi-asserted-by":"publisher","first-page":"e79925","DOI":"10.7554\/eLife.79925","article-title":"Lineage-specific differences and regulatory networks governing human chondrocyte development","volume":"12","author":"Richard","year":"2023","journal-title":"elife"},{"issue":"7948","key":"2025010523412045100_ref84","doi-asserted-by":"publisher","first-page":"509","DOI":"10.1038\/s41586-022-05649-2","article-title":"Reconstituting human somitogenesis in vitro","volume":"614","author":"Yamanaka","year":"2023","journal-title":"Nature"},{"issue":"7948","key":"2025010523412045100_ref85","doi-asserted-by":"publisher","first-page":"500","DOI":"10.1038\/s41586-022-05655-4","article-title":"Reconstruction and deconstruction of human somitogenesis in vitro","volume":"614","author":"Miao","year":"2023","journal-title":"Nature"},{"key":"2025010523412045100_ref86","doi-asserted-by":"publisher","first-page":"536","DOI":"10.1016\/j.reth.2023.09.017","article-title":"Modeling of intramembranous ossification using human pluripotent stem cell-derived paraxial mesoderm derivatives","volume":"24","author":"Ikeda","year":"2023","journal-title":"Regen Ther"},{"issue":"4","key":"2025010523412045100_ref87","doi-asserted-by":"publisher","first-page":"207","DOI":"10.1038\/s41576-018-0089-8","article-title":"Chromatin accessibility and the regulatory epigenome","volume":"20","author":"Klemm","year":"2019","journal-title":"Nat Rev Genet"},{"issue":"5","key":"2025010523412045100_ref88","doi-asserted-by":"publisher","first-page":"927","DOI":"10.1016\/j.devcel.2012.03.011","article-title":"FoxA family members are crucial regulators of the hypertrophic chondrocyte differentiation program","volume":"22","author":"Ionescu","year":"2012","journal-title":"Dev Cell"},{"issue":"3","key":"2025010523412045100_ref89","doi-asserted-by":"crossref","first-page":"238","DOI":"10.1016\/j.devcel.2016.04.002","article-title":"Sp7\/Osterix is restricted to bone-forming vertebrates where it acts as a dlx Co-factor in osteoblast specification","volume":"37","author":"Hojo","year":"2016","journal-title":"Dev Cell"},{"issue":"10","key":"2025010523412045100_ref90","doi-asserted-by":"publisher","first-page":"111315","DOI":"10.1016\/j.celrep.2022.111315","article-title":"Runx2 regulates chromatin accessibility to direct the osteoblast program at neonatal stages","volume":"40","author":"Hojo","year":"2022","journal-title":"Cell Rep"},{"issue":"12","key":"2025010523412045100_ref91","doi-asserted-by":"publisher","first-page":"e1004820","DOI":"10.1371\/journal.pgen.1004820","article-title":"Chondrocytes transdifferentiate into osteoblasts in endochondral bone during development, postnatal growth and fracture healing in mice","volume":"10","author":"Zhou","year":"2014","journal-title":"PLoS Genet"},{"issue":"8","key":"2025010523412045100_ref92","doi-asserted-by":"publisher","first-page":"926","DOI":"10.1038\/s41551-021-00778-x","article-title":"Induction and expansion of human PRRX1+ limb-bud-like mesenchymal cells from pluripotent stem cells","volume":"5","author":"Yamada","year":"2021","journal-title":"Nature Biomedical Engineering"},{"issue":"4","key":"2025010523412045100_ref93","doi-asserted-by":"publisher","first-page":"101786","DOI":"10.1016\/j.xpro.2022.101786","article-title":"A protocol to induce expandable limb-bud mesenchymal cells from human pluripotent stem cells","volume":"3","author":"Takao","year":"2022","journal-title":"STAR Protocols"},{"issue":"1","key":"2025010523412045100_ref94","doi-asserted-by":"crossref","first-page":"3802","DOI":"10.1038\/s41467-019-11702-y","article-title":"Self-organized formation of developing appendages from murine pluripotent stem cells","volume":"10","author":"Mori","year":"2019","journal-title":"Nat Commun"},{"issue":"7","key":"2025010523412045100_ref95","doi-asserted-by":"publisher","first-page":"923","DOI":"10.1634\/stemcells.2005-0038","article-title":"Generation of peripheral sensory and sympathetic neurons and neural crest cells from human embryonic stem cells","volume":"23","author":"Pomp","year":"2005","journal-title":"Stem cells (Dayton, Ohio)"},{"issue":"2","key":"2025010523412045100_ref96","doi-asserted-by":"publisher","first-page":"145","DOI":"10.1111\/j.1432-0436.2007.00196.x","article-title":"Peripheral sensory neurons differentiate from neural precursors derived from human embryonic stem cells","volume":"76","author":"Brokhman","year":"2008","journal-title":"Differentiation"},{"issue":"3","key":"2025010523412045100_ref97","doi-asserted-by":"publisher","first-page":"275","DOI":"10.1038\/nbt.1529","article-title":"Highly efficient neural conversion of human ES and iPS cells by dual inhibition of SMAD signaling","volume":"27","author":"Chambers","year":"2009","journal-title":"Nat Biotechnol"},{"issue":"7283","key":"2025010523412045100_ref98","doi-asserted-by":"publisher","first-page":"958","DOI":"10.1038\/nature08733","article-title":"CHD7 cooperates with PBAF to control multipotent neural crest formation","volume":"463","author":"Bajpai","year":"2010","journal-title":"Nature"},{"issue":"4","key":"2025010523412045100_ref99","doi-asserted-by":"publisher","first-page":"688","DOI":"10.1038\/nprot.2010.35","article-title":"Derivation of neural crest cells from human pluripotent stem cells","volume":"5","author":"Lee","year":"2010","journal-title":"Nat Protoc"},{"issue":"48","key":"2025010523412045100_ref100","doi-asserted-by":"publisher","first-page":"19240","DOI":"10.1073\/pnas.1113746108","article-title":"Wnt signaling and a Smad pathway blockade direct the differentiation of human pluripotent stem cells to multipotent neural crest cells","volume":"108","author":"Menendez","year":"2011","journal-title":"Proc Natl Acad Sci"},{"issue":"3","key":"2025010523412045100_ref101","doi-asserted-by":"publisher","first-page":"398","DOI":"10.1242\/dev.130849","article-title":"WNT\/\u03b2-catenin signaling mediates human neural crest induction via a pre-neural border intermediate","volume":"143","author":"Leung","year":"2016","journal-title":"Development"},{"issue":"1","key":"2025010523412045100_ref102","doi-asserted-by":"publisher","first-page":"203","DOI":"10.1038\/nprot.2012.156","article-title":"Directed differentiation of human pluripotent cells to neural crest stem cells","volume":"8","author":"Menendez","year":"2013","journal-title":"Nat Protoc"},{"issue":"12","key":"2025010523412045100_ref103","doi-asserted-by":"publisher","first-page":"e112291","DOI":"10.1371\/journal.pone.0112291","article-title":"Derivation of mesenchymal stromal cells from pluripotent stem cells through a neural crest lineage using small molecule compounds with defined media","volume":"9","author":"Fukuta","year":"2014","journal-title":"PLoS One"},{"key":"2025010523412045100_ref104","doi-asserted-by":"publisher","first-page":"290","DOI":"10.1016\/j.bioactmat.2021.11.014","article-title":"iPSC-derived cranial neural crest-like cells can replicate dental pulp tissue with the aid of angiogenic hydrogel","volume":"14","author":"Kobayashi","year":"2022","journal-title":"Bioactive Materials"},{"key":"2025010523412045100_ref105","doi-asserted-by":"publisher","first-page":"25","DOI":"10.1016\/j.reth.2019.04.004","article-title":"Insights into the biology of fibrodysplasia ossificans progressiva using patient-derived induced pluripotent stem cells","volume":"11","author":"Nakajima","year":"2019","journal-title":"Regenerative Therapy"},{"issue":"1","key":"2025010523412045100_ref106","doi-asserted-by":"publisher","first-page":"190","DOI":"10.1186\/1750-1172-8-190","article-title":"Induced pluripotent stem cells from patients with human fibrodysplasia ossificans progressiva show increased mineralization and cartilage formation","volume":"8","author":"Matsumoto","year":"2013","journal-title":"Orphanet J Rare Dis"},{"issue":"50","key":"2025010523412045100_ref107","doi-asserted-by":"publisher","first-page":"15438","DOI":"10.1073\/pnas.1510540112","article-title":"Neofunction of ACVR1 in fibrodysplasia ossificans progressiva","volume":"112","author":"Hino","year":"2015","journal-title":"Proc Natl Acad Sci"},{"issue":"9","key":"2025010523412045100_ref108","doi-asserted-by":"publisher","first-page":"3339","DOI":"10.1172\/JCI93521","article-title":"Activin-a enhances mTOR signaling to promote aberrant chondrogenesis in fibrodysplasia ossificans progressiva","volume":"127","author":"Hino","year":"2017","journal-title":"J Clin Invest"},{"key":"2025010523412045100_ref109","doi-asserted-by":"publisher","first-page":"100027","DOI":"10.1074\/jbc.RA120.014709","article-title":"4-Phenylbutyric acid enhances the mineralization of osteogenesis imperfecta iPSC-derived osteoblasts","volume":"296","author":"Takeyari","year":"2021","journal-title":"J Biol Chem"},{"issue":"1","key":"2025010523412045100_ref110","doi-asserted-by":"publisher","first-page":"12","DOI":"10.1186\/s13287-017-0754-4","article-title":"Targeted reversion of induced pluripotent stem cells from patients with human cleidocranial dysplasia improves bone regeneration in a rat calvarial bone defect model","volume":"9","author":"Saito","year":"2018","journal-title":"Stem Cell Res Ther"},{"issue":"10","key":"2025010523412045100_ref111","doi-asserted-by":"publisher","first-page":"e0186879","DOI":"10.1371\/journal.pone.0186879","article-title":"Gorlin syndrome-derived induced pluripotent stem cells are hypersensitive to hedgehog-mediated osteogenic induction","volume":"12","author":"Hasegawa","year":"2017","journal-title":"PLoS One"},{"issue":"7519","key":"2025010523412045100_ref112","doi-asserted-by":"publisher","first-page":"507","DOI":"10.1038\/nature13775","article-title":"Statin treatment rescues FGFR3 skeletal dysplasia phenotypes","volume":"513","author":"Yamashita","year":"2014","journal-title":"Nature"},{"issue":"3","key":"2025010523412045100_ref113","doi-asserted-by":"publisher","first-page":"610","DOI":"10.1016\/j.stemcr.2021.01.014","article-title":"Differentiation of hypertrophic chondrocytes from human iPSCs for the In vitro Modeling of chondrodysplasias","volume":"16","author":"Pretemer","year":"2021","journal-title":"Stem cell reports"},{"issue":"2","key":"2025010523412045100_ref114","doi-asserted-by":"publisher","first-page":"299","DOI":"10.1093\/hmg\/ddu444","article-title":"Modeling type II collagenopathy skeletal dysplasia by directed conversion and induced pluripotent stem cells","volume":"24","author":"Okada","year":"2014","journal-title":"Hum Mol Genet"},{"key":"2025010523412045100_ref115","doi-asserted-by":"publisher","first-page":"101962","DOI":"10.1016\/j.scr.2020.101962","article-title":"CRISPR\/Cas9 editing to generate a heterozygous COL2A1 p.G1170S human chondrodysplasia iPSC line, MCRIi019-a-2, in a control iPSC line, MCRIi019-a","volume":"48","author":"Kung","year":"2020","journal-title":"Stem Cell Res"},{"issue":"9","key":"2025010523412045100_ref116","doi-asserted-by":"publisher","first-page":"1171","DOI":"10.5966\/sctm.2015-0384","article-title":"Chondrocytes derived from mesenchymal stromal cells and induced pluripotent cells of patients with familial Osteochondritis Dissecans exhibit an endoplasmic reticulum stress response and defective matrix assembly","volume":"5","author":"Xu","year":"2016","journal-title":"Stem Cells Transl Med"},{"issue":"5","key":"2025010523412045100_ref117","doi-asserted-by":"publisher","first-page":"293","DOI":"10.1093\/stcltm\/szad018","article-title":"Patient-specific iPSC-derived models link aberrant endoplasmic reticulum stress sensing and response to juvenile Osteochondritis Dissecans Etiology","volume":"12","author":"Salazar-Noratto","year":"2023","journal-title":"Stem Cells Transl Med"},{"issue":"4","key":"2025010523412045100_ref118","doi-asserted-by":"publisher","first-page":"321","DOI":"10.1016\/j.jare.2017.02.004","article-title":"iPS cell technologies and their prospect for bone regeneration and disease modeling: a mini review","volume":"8","author":"Csobonyeiova","year":"2017","journal-title":"J Adv Res"},{"issue":"1","key":"2025010523412045100_ref119","doi-asserted-by":"publisher","first-page":"126","DOI":"10.1038\/s41392-023-01338-2","article-title":"Characterization of mesenchymal stem cells in human fetal bone marrow by single-cell transcriptomic and functional analysis. Signal transduction and targeted","volume":"8","author":"Zhang","year":"2023","journal-title":"Therapy"},{"key":"2025010523412045100_ref120","doi-asserted-by":"publisher","first-page":"e81656","DOI":"10.7554\/eLife.81656","article-title":"Identification of phenotypically, functionally, and anatomically distinct stromal niche populations in human bone marrow based on single-cell RNA sequencing","volume":"12","author":"Li","year":"2023","journal-title":"elife"},{"issue":"17","key":"2025010523412045100_ref121","doi-asserted-by":"publisher","first-page":"3706","DOI":"10.1016\/j.cell.2023.07.009","article-title":"Distinct molecular profiles of skull bone marrow in health and neurological disorders","volume":"186","author":"Kolabas","year":"2023","journal-title":"Cell"},{"issue":"7755","key":"2025010523412045100_ref122","doi-asserted-by":"publisher","first-page":"222","DOI":"10.1038\/s41586-019-1104-8","article-title":"The bone marrow microenvironment at single-cell resolution","volume":"569","author":"Tikhonova","year":"2019","journal-title":"Nature"},{"issue":"10","key":"2025010523412045100_ref123","doi-asserted-by":"publisher","first-page":"1981","DOI":"10.1002\/jbmr.4052","article-title":"Single-cell RNA sequencing of Calvarial and long-bone Endocortical cells","volume":"35","author":"Ayturk","year":"2020","journal-title":"Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research"},{"issue":"1","key":"2025010523412045100_ref124","doi-asserted-by":"publisher","first-page":"51","DOI":"10.1038\/nrg.2016.138","article-title":"From profiles to function in epigenomics","volume":"18","author":"Stricker","year":"2017","journal-title":"Nat Rev Genet"},{"issue":"8","key":"2025010523412045100_ref125","doi-asserted-by":"publisher","first-page":"467","DOI":"10.1038\/s41576-022-00466-9","article-title":"Human organs-on-chips for disease modelling, drug development and personalized medicine","volume":"23","author":"Ingber","year":"2022","journal-title":"Nat Rev Genet"},{"issue":"18","key":"2025010523412045100_ref126","doi-asserted-by":"crossref","first-page":"4597","DOI":"10.1016\/j.cell.2021.08.005","article-title":"Organs-on-a-chip models for biological research","volume":"184","author":"Vunjak-Novakovic","year":"2021","journal-title":"Cell"},{"issue":"6","key":"2025010523412045100_ref127","doi-asserted-by":"publisher","first-page":"3058","DOI":"10.1021\/acsbiomaterials.3c00066","article-title":"Bone-on-a-Chip: biomimetic models based on microfluidic Technologies for Biomedical Applications","volume":"9","author":"Kim","year":"2023","journal-title":"ACS Biomaterials Science & Engineering"},{"issue":"6","key":"2025010523412045100_ref128","doi-asserted-by":"publisher","first-page":"993","DOI":"10.1016\/j.stem.2021.05.008","article-title":"Harnessing organs-on-a-chip to model tissue regeneration","volume":"28","author":"Tavakol","year":"2021","journal-title":"Cell Stem Cell"},{"issue":"1","key":"2025010523412045100_ref129","doi-asserted-by":"publisher","first-page":"299","DOI":"10.1038\/s42003-023-04694-5","article-title":"Gradient to sectioning CUBE workflow for the generation and imaging of organoids with localized differentiation","volume":"6","author":"Koh","year":"2023","journal-title":"Communications Biology"},{"issue":"6","key":"2025010523412045100_ref130","doi-asserted-by":"publisher","first-page":"546","DOI":"10.1093\/stmcls\/sxac020","article-title":"Human induced pluripotent stem cells: from cell origin, genomic stability, and epigenetic memory to translational medicine","volume":"40","author":"Poetsch","year":"2022","journal-title":"Stem cells (Dayton, Ohio)"},{"issue":"4","key":"2025010523412045100_ref131","doi-asserted-by":"publisher","first-page":"518","DOI":"10.1016\/j.stem.2016.11.005","article-title":"Analysis of transcriptional variability in a large human iPSC library reveals genetic and non-genetic determinants of heterogeneity","volume":"20","author":"Carcamo-Orive","year":"2017","journal-title":"Cell Stem Cell"},{"issue":"1","key":"2025010523412045100_ref132","doi-asserted-by":"publisher","DOI":"10.1242\/dmm.042317","article-title":"Addressing variability in iPSC-derived models of human disease: guidelines to promote reproducibility","volume":"13","author":"Volpato","year":"2020","journal-title":"Dis Model Mech"},{"key":"2025010523412045100_ref133","doi-asserted-by":"publisher","first-page":"e83138","DOI":"10.7554\/eLife.83138","article-title":"Differential chondrogenic differentiation between iPSC derived from healthy and OA cartilage is associated with changes in epigenetic regulation and metabolic transcriptomic signatures","volume":"12","author":"Khan","year":"2023","journal-title":"elife"},{"issue":"1","key":"2025010523412045100_ref134","doi-asserted-by":"publisher","first-page":"4272","DOI":"10.1038\/s41598-020-61071-6","article-title":"Generation and characterization of human induced pluripotent stem cells (iPSCs) from hand osteoarthritis patient-derived fibroblasts","volume":"10","author":"Castro-Vi\u00f1uelas","year":"2020","journal-title":"Sci Rep"},{"issue":"10","key":"2025010523412045100_ref135","doi-asserted-by":"publisher","first-page":"1475","DOI":"10.1016\/j.stem.2022.09.008","article-title":"A human iPSC-array-based GWAS identifies a virus susceptibility locus in the NDUFA4 gene and functional variants","volume":"29","author":"Han","year":"2022","journal-title":"Cell Stem Cell"},{"issue":"1","key":"2025010523412045100_ref136","doi-asserted-by":"publisher","first-page":"160","DOI":"10.1016\/j.stem.2021.11.001","article-title":"Genome-wide screening in human kidney organoids identifies developmental and disease-related aspects of nephrogenesis","volume":"29","author":"Ungricht","year":"2022","journal-title":"Cell Stem Cell"},{"issue":"5","key":"2025010523412045100_ref137","doi-asserted-by":"publisher","first-page":"402","DOI":"10.1038\/s41578-021-00279-y","article-title":"Engineering organoids","volume":"6","author":"Hofer","year":"2021","journal-title":"Nature Reviews Materials"}],"container-title":["Journal of Bone and Mineral Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/jbmr\/advance-article-pdf\/doi\/10.1093\/jbmr\/zjae178\/60961413\/zjae178.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jbmr\/article-pdf\/40\/1\/5\/60961413\/zjae178.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jbmr\/article-pdf\/40\/1\/5\/60961413\/zjae178.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,1,6]],"date-time":"2025-01-06T04:29:42Z","timestamp":1736137782000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jbmr\/article\/40\/1\/5\/7875970"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,11,5]]},"references-count":137,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2024,11,5]]},"published-print":{"date-parts":[[2024,12,31]]}},"URL":"https:\/\/doi.org\/10.1093\/jbmr\/zjae178","relation":{},"ISSN":["0884-0431","1523-4681"],"issn-type":[{"value":"0884-0431","type":"print"},{"value":"1523-4681","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2025,1]]},"published":{"date-parts":[[2024,11,5]]}}}