{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,21]],"date-time":"2026-10-21T12:45:22Z","timestamp":1792586722124,"version":"4.5.0"},"reference-count":31,"publisher":"MyJove Corporation","issue":"233","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JoVE"],"DOI":"10.3791\/68556","type":"journal-article","created":{"date-parts":[[2026,7,28]],"date-time":"2026-07-28T23:02:46Z","timestamp":1785279766000},"source":"Crossref","is-referenced-by-count":0,"title":["Patient-Specific iPSC-Derived Cornea Organoids for Investigating Aniridia-Associated Corneal Disorders"],"prefix":"10.3791","author":[{"given":"Ali Can","family":"Ko\u00e7","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gaye Su","family":"Yi\u011fit","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Semih Numan","family":"G\u00f6\u00e7mez","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mustafa","family":"Titiz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sinan","family":"G\u00fcven","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"2098","published-online":{"date-parts":[[2026,7,28]]},"reference":[{"issue":"3","key":"ref1","doi-asserted-by":"crossref","first-page":"326","DOI":"10.1016\/j.exer.2010.06.021","type":"journal-article","article-title":"The molecular basis of corneal transparency","volume":"91","author":"Hassell","year":"2010","unstructured":"Hassell, J. R., Birk, D. E. The molecular basis of corneal transparency. Exp Eye Res. 91 (3), 326-335 (2010).","journal-title":"Exp Eye Res"},{"key":"ref2","doi-asserted-by":"crossref","first-page":"101133","DOI":"10.1016\/j.preteyeres.2022.101133","type":"journal-article","article-title":"Congenital aniridia beyond black eyes: From phenotype and novel genetic mechanisms to innovative therapeutic approaches","volume":"95","author":"Daruich","year":"2023","unstructured":"Daruich, A. et al. Congenital aniridia beyond black eyes: From phenotype and novel genetic mechanisms to innovative therapeutic approaches. Prog Retin Eye Res. 95, 101133 (2023).","journal-title":"Prog Retin Eye Res"},{"issue":"10","key":"ref3","doi-asserted-by":"crossref","first-page":"1096","DOI":"10.1097\/ICO.0b013e3181d20493","type":"journal-article","article-title":"Corneal involvement in congenital aniridia","volume":"29","author":"Eden","year":"2010","unstructured":"Eden, U., Riise, R., Tornqvist, K. Corneal involvement in congenital aniridia. Cornea. 29 (10), 1096-1102 (2010).","journal-title":"Cornea"},{"issue":"18","key":"ref4","doi-asserted-by":"crossref","first-page":"3113","DOI":"10.1242\/dev.118570","type":"journal-article","article-title":"Modeling mouse and human development using organoid cultures","volume":"142","author":"Huch","year":"2015","unstructured":"Huch, M., Koo, B. K. Modeling mouse and human development using organoid cultures. Development. 142 (18), 3113-3125 (2015).","journal-title":"Development"},{"issue":"5","key":"ref5","doi-asserted-by":"crossref","first-page":"402","DOI":"10.1038\/s41578-021-00279-y","type":"journal-article","article-title":"Engineering organoids","volume":"6","author":"Hofer","year":"2021","unstructured":"Hofer, M., Lutolf, M. P. Engineering organoids. Nat Rev Mater. 6 (5), 402-420 (2021).","journal-title":"Nat Rev Mater"},{"issue":"1","key":"ref6","doi-asserted-by":"crossref","first-page":"25","DOI":"10.1016\/j.stem.2015.12.005","type":"journal-article","article-title":"Engineering Stem Cell Organoids","volume":"18","author":"Yin","year":"2016","unstructured":"Yin, X. et al. Engineering Stem Cell Organoids. Cell Stem Cell. 18 (1), 25-38 (2016).","journal-title":"Cell Stem Cell"},{"key":"ref7","doi-asserted-by":"crossref","first-page":"41286","DOI":"10.1038\/srep41286","type":"journal-article","article-title":"Cornea organoids from human induced pluripotent stem cells","volume":"7","author":"Foster","year":"2017","unstructured":"Foster, J. W. et al. Cornea organoids from human induced pluripotent stem cells. Sci Rep. 7, 41286 (2017).","journal-title":"Sci Rep"},{"issue":"13","key":"ref8","first-page":"2338","type":"journal-article","article-title":"Generating minicorneal organoids from human induced pluripotent stem cells","volume":"144","author":"Susaimanickam","year":"2017","unstructured":"Susaimanickam, P. J. et al. Generating minicorneal organoids from human induced pluripotent stem cells. Development. 144 (13), 2338-2351 (2017).","journal-title":"Development"},{"issue":"1","key":"ref9","doi-asserted-by":"crossref","first-page":"14","DOI":"10.1186\/s12886-024-03831-w","type":"journal-article","article-title":"Patient-derived cornea organoid model to study metabolomic characterization of rare disease: aniridia-associated keratopathy","volume":"25","author":"Koc","year":"2025","unstructured":"Koc, A. C. et al. Patient-derived cornea organoid model to study metabolomic characterization of rare disease: aniridia-associated keratopathy. BMC Ophthalmol. 25 (1), 14 (2025).","journal-title":"BMC Ophthalmol"},{"issue":"12","key":"ref10","doi-asserted-by":"crossref","first-page":"1133","DOI":"10.1093\/stmcls\/sxad067","type":"journal-article","article-title":"Production and Limbal Lineage Commitment of Aniridia Patient-Derived Induced Pluripotent Stem Cells","volume":"41","author":"Ilmarinen","year":"2023","unstructured":"Ilmarinen, T. et al. Production and Limbal Lineage Commitment of Aniridia Patient-Derived Induced Pluripotent Stem Cells. Stem Cells. 41 (12), 1133-1141 (2023).","journal-title":"Stem Cells"},{"issue":"7173","key":"ref11","doi-asserted-by":"crossref","first-page":"1230","DOI":"10.1038\/nature06403","type":"journal-article","article-title":"Nanog safeguards pluripotency and mediates germline development","volume":"450","author":"Chambers","year":"2007","unstructured":"Chambers, I. et al. Nanog safeguards pluripotency and mediates germline development. Nature. 450 (7173), 1230-1234 (2007).","journal-title":"Nature"},{"issue":"1","key":"ref12","doi-asserted-by":"crossref","first-page":"211","DOI":"10.1186\/s13287-019-1316-8","type":"journal-article","article-title":"Osteopetrotic induced pluripotent stem cells derived from patients with different disease-associated mutations by non-integrating reprogramming methods","volume":"10","author":"Okur","year":"2019","unstructured":"Okur, F. V., Cevher, I., Ozdemir, C., Kocaefe, C., Cetinkaya, D. U. Osteopetrotic induced pluripotent stem cells derived from patients with different disease-associated mutations by non-integrating reprogramming methods. Stem Cell Res Ther. 10 (1), 211 (2019).","journal-title":"Stem Cell Res Ther"},{"issue":"6","key":"ref13","doi-asserted-by":"crossref","first-page":"771","DOI":"10.1016\/j.stem.2012.05.009","type":"journal-article","article-title":"Self-formation of optic cups and storable stratified neural retina from human ESCs","volume":"10","author":"Nakano","year":"2012","unstructured":"Nakano, T. et al. Self-formation of optic cups and storable stratified neural retina from human ESCs. Cell Stem Cell. 10 (6), 771-785 (2012).","journal-title":"Cell Stem Cell"},{"issue":"12","key":"ref14","doi-asserted-by":"crossref","first-page":"1050","DOI":"10.3390\/genes10121050","type":"journal-article","article-title":"The Spectrum of PAX6 Mutations and Genotype-Phenotype Correlations in the Eye","volume":"10","author":"Lima Cunha","year":"2019","unstructured":"Lima Cunha, D., Arno, G., Corton, M., Moosajee, M. The Spectrum of PAX6 Mutations and Genotype-Phenotype Correlations in the Eye. Genes (Basel). 10 (12), 1050 (2019).","journal-title":"Genes (Basel)"},{"issue":"7594","key":"ref15","doi-asserted-by":"crossref","first-page":"376","DOI":"10.1038\/nature17000","type":"journal-article","article-title":"Co-ordinated ocular development from human iPS cells and recovery of corneal function","volume":"531","author":"Hayashi","year":"2016","unstructured":"Hayashi, R. et al. Co-ordinated ocular development from human iPS cells and recovery of corneal function. Nature. 531 (7594), 376-380 (2016).","journal-title":"Nature"},{"issue":"2","key":"ref16","doi-asserted-by":"crossref","DOI":"10.1002\/wdev.303","type":"journal-article","article-title":"Limbal stem cells: identity, developmental origin, and therapeutic potential","volume":"7","author":"Gonzalez","year":"2018","unstructured":"Gonzalez, G., Sasamoto, Y., Ksander, B. R., Frank, M. H., Frank, N. Y. Limbal stem cells: identity, developmental origin, and therapeutic potential. Wiley Interdiscip Rev Dev Biol. 7 (2), 10.1002\/wdev.303 (2018).","journal-title":"Wiley Interdiscip Rev Dev Biol"},{"issue":"1","key":"ref17","doi-asserted-by":"crossref","first-page":"11374","DOI":"10.1038\/s41598-017-11112-4","type":"journal-article","article-title":"MicroRNA-28 potentially regulates the photoreceptor lineage commitment of Muller glia-derived progenitors","volume":"7","author":"Ji","year":"2017","unstructured":"Ji, H. P. et al. MicroRNA-28 potentially regulates the photoreceptor lineage commitment of Muller glia-derived progenitors. Sci Rep. 7 (1), 11374 (2017).","journal-title":"Sci Rep"},{"issue":"8","key":"ref18","doi-asserted-by":"crossref","first-page":"3935","DOI":"10.1167\/iovs.09-4027","type":"journal-article","article-title":"Role of insulin in regulation of Na+-\/K+-dependent ATPase activity and pump function in corneal endothelial cells","volume":"51","author":"Hatou","year":"2010","unstructured":"Hatou, S. et al. Role of insulin in regulation of Na+-\/K+-dependent ATPase activity and pump function in corneal endothelial cells. Invest Ophthalmol Vis Sci. 51 (8), 3935-3942 (2010).","journal-title":"Invest Ophthalmol Vis Sci"},{"issue":"2","key":"ref19","doi-asserted-by":"crossref","first-page":"147","DOI":"10.1016\/j.scr.2012.11.001","type":"journal-article","article-title":"Aquaporin 1-positive stromal niche-like cells directly interact with N-cadherin-positive clusters in the basal limbal epithelium","volume":"10","author":"Higa","year":"2013","unstructured":"Higa, K. et al. Aquaporin 1-positive stromal niche-like cells directly interact with N-cadherin-positive clusters in the basal limbal epithelium. Stem Cell Res. 10 (2), 147-155 (2013).","journal-title":"Stem Cell Res"},{"key":"ref20","doi-asserted-by":"crossref","first-page":"27462","DOI":"10.1038\/srep27462","type":"journal-article","article-title":"Stromal fibroblasts derived from mammary gland of bovine with mastitis display inflammation-specific changes","volume":"6","author":"Chen","year":"2016","unstructured":"Chen, Q. et al. Stromal fibroblasts derived from mammary gland of bovine with mastitis display inflammation-specific changes. Sci Rep. 6, 27462 (2016).","journal-title":"Sci Rep"},{"key":"ref21","doi-asserted-by":"crossref","first-page":"252","DOI":"10.2174\/1874364101711010252","type":"journal-article","article-title":"Pathophysiology of Keratoconus: What Do We Know Today","volume":"11","author":"Soiberman","year":"2017","unstructured":"Soiberman, U., Foster, J. W., Jun, A. S., Chakravarti, S. Pathophysiology of Keratoconus: What Do We Know Today. Open Ophthalmol J. 11, 252-261 (2017).","journal-title":"Open Ophthalmol J"},{"key":"ref22","doi-asserted-by":"crossref","first-page":"240","DOI":"10.1016\/j.omtn.2023.06.016","type":"journal-article","article-title":"Restoration of functional PAX6 in aniridia patient iPSC-derived ocular tissue models using repurposed nonsense suppression drugs","volume":"33","author":"Lima Cunha","year":"2023","unstructured":"Lima Cunha, D. et al. Restoration of functional PAX6 in aniridia patient iPSC-derived ocular tissue models using repurposed nonsense suppression drugs. Mol Ther Nucleic Acids. 33, 240-253 (2023).","journal-title":"Mol Ther Nucleic Acids"},{"issue":"6","key":"ref23","doi-asserted-by":"crossref","first-page":"167869","DOI":"10.1016\/j.bbadis.2025.167869","type":"journal-article","article-title":"Variant-specific disruption to notch signalling in PAX6 microphthalmia and aniridia patient-derived hiPSC optic cup-like organoids","volume":"1871","author":"Harding","year":"2025","unstructured":"Harding, P. et al. Variant-specific disruption to notch signalling in PAX6 microphthalmia and aniridia patient-derived hiPSC optic cup-like organoids. Biochim Biophys Acta Mol Basis Dis. 1871 (6), 167869 (2025).","journal-title":"Biochim Biophys Acta Mol Basis Dis"},{"issue":"8","key":"ref24","first-page":"2821","type":"journal-article","article-title":"Transcriptomic analysis of aniridia iPSC-derived limbal epithelial stem cells reveals early changes linked to the limbal niche","volume":"64","author":"Cunha","year":"2023","unstructured":"Cunha, D. L., Smits, J., Zhou, H. Q., Moosajee, M. Transcriptomic analysis of aniridia iPSC-derived limbal epithelial stem cells reveals early changes linked to the limbal niche. Invest Ophthalmol Vis Sci. 64 (8), 2821 (2023).","journal-title":"Invest Ophthalmol Vis Sci"},{"key":"ref25","doi-asserted-by":"crossref","DOI":"10.1016\/j.scr.2021.102184","type":"journal-article","article-title":"Generation of human iPSC line (UCLi013-A) from a patient with microphthalmia and aniridia, carrying a heterozygous missense mutation c.372C>A p.(Asn124Lys) in","volume":"51","author":"Harding","year":"2021","unstructured":"Harding, P. et al. Generation of human iPSC line (UCLi013-A) from a patient with microphthalmia and aniridia, carrying a heterozygous missense mutation c.372C>A p.(Asn124Lys) in. Stem Cell Res. 51, 10.1016\/j.scr.2021.102184 (2021).","journal-title":"Stem Cell Res"},{"issue":"21","key":"ref26","doi-asserted-by":"crossref","DOI":"10.3390\/ijms21217834","type":"journal-article","article-title":"Differentiation Induction of Human Stem Cells for Corneal Epithelial Regeneration","volume":"21","author":"Theerakittayakorn","year":"2020","unstructured":"Theerakittayakorn, K. et al. Differentiation Induction of Human Stem Cells for Corneal Epithelial Regeneration. Int J Mol Sci. 21 (21), 10.3390\/ijms21217834, (2020).","journal-title":"Int J Mol Sci"},{"issue":"11","key":"ref27","doi-asserted-by":"crossref","first-page":"1170","DOI":"10.1038\/nmeth.3142","type":"journal-article","article-title":"Small molecules facilitate rapid and synchronous iPSC generation","volume":"11","author":"Bar-Nur","year":"2014","unstructured":"Bar-Nur, O. et al. Small molecules facilitate rapid and synchronous iPSC generation. Nat Methods. 11 (11), 1170-1176 (2014).","journal-title":"Nat Methods"},{"issue":"5","key":"ref28","doi-asserted-by":"crossref","first-page":"pgac246","DOI":"10.1093\/pnasnexus\/pgac246","type":"journal-article","article-title":"Single cell RNA-seq of human cornea organoids identifies cell fates of a developing immature cornea","volume":"1","author":"Maiti","year":"2022","unstructured":"Maiti, G. et al. Single cell RNA-seq of human cornea organoids identifies cell fates of a developing immature cornea. PNAS Nexus. 1 (5), pgac246 (2022).","journal-title":"PNAS Nexus"},{"key":"ref29","doi-asserted-by":"crossref","first-page":"140","DOI":"10.1016\/j.jtos.2021.12.003","type":"journal-article","article-title":"The antipsychotropic drug Duloxetine rescues PAX6 haploinsufficiency of mutant limbal stem cells through inhibition of the MEK\/ERK signaling pathway","volume":"23","author":"Dorot","year":"2022","unstructured":"Dorot, O. et al. The antipsychotropic drug Duloxetine rescues PAX6 haploinsufficiency of mutant limbal stem cells through inhibition of the MEK\/ERK signaling pathway. Ocul Surf. 23, 140-142 (2022).","journal-title":"Ocul Surf"},{"issue":"5","key":"ref30","doi-asserted-by":"crossref","first-page":"940","DOI":"10.1016\/j.survophthal.2023.04.003","type":"journal-article","article-title":"Future directions in managing aniridia-associated keratopathy","volume":"68","author":"van Velthoven","year":"2023","unstructured":"van Velthoven, A. J. H. et al. Future directions in managing aniridia-associated keratopathy. Surv Ophthalmol. 68 (5), 940-956 (2023).","journal-title":"Surv Ophthalmol"},{"issue":"5","key":"ref31","doi-asserted-by":"crossref","first-page":"e2400018","DOI":"10.1002\/adbi.202400018","type":"journal-article","article-title":"Generation of Anterior Segment of the Eye Cells from hiPSCs in Microfluidic Platforms","volume":"8","author":"Kocak","year":"2024","unstructured":"Kocak, G. et al. Generation of Anterior Segment of the Eye Cells from hiPSCs in Microfluidic Platforms. Adv Biol (Weinh). 8 (5), e2400018 (2024).","journal-title":"Adv Biol (Weinh)"}],"container-title":["Journal of Visualized Experiments"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.jove.com\/t\/68556\/patient-specific-ipsc-derived-cornea-organoids-for-investigating","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,10,21]],"date-time":"2026-10-21T12:16:13Z","timestamp":1792584973000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.jove.com\/t\/68556\/patient-specific-ipsc-derived-cornea-organoids-for-investigating"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,7,28]]},"references-count":31,"journal-issue":{"issue":"233","published-online":{"date-parts":[[2026]]}},"URL":"https:\/\/doi.org\/10.3791\/68556","relation":{},"ISSN":["1940-087X"],"issn-type":[{"value":"1940-087X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,7,28]]}}}