{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,22]],"date-time":"2026-01-22T01:58:10Z","timestamp":1769047090098,"version":"3.49.0"},"reference-count":89,"publisher":"The Company of Biologists","issue":"8","license":[{"start":{"date-parts":[[2023,8,29]],"date-time":"2023-08-29T00:00:00Z","timestamp":1693267200000},"content-version":"vor","delay-in-days":28,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["82070984"],"award-info":[{"award-number":["82070984"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["82271097"],"award-info":[{"award-number":["82271097"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["82101109"],"award-info":[{"award-number":["82101109"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100007835","name":"Wenzhou Medical University","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100007835","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,8,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Hedgehog (Hh) signaling is well known for its crucial role during development, but its specific role in individual cell lineages is less well characterized. Here, we disrupted Hh signaling specifically in melanocytes by using Cre-mediated cell-type-specific knockout of the Hh regulator suppressor of fused (Sufu). Interestingly, corresponding mice were fully pigmented and showed no developmental alterations in melanocyte numbers or distribution in skin and hair follicles. However, there were ectopic melanoblasts visible in the anterior chamber of the eye that eventually displayed severe malformation. Choroidal melanocytes remained unaltered. Surprisingly, the abnormal accumulation of anterior uveal melanoblasts was not the result of increased cell proliferation but of increased migration to ectopic locations such as the cornea. In melanoblasts in vitro, Sufu knockdown replicated the increase in cell migration without affecting proliferation and was mediated by an increased level of phosphorylated-ERK brought about by a reduction in the levels of the repressor form of GLI3. These results highlight the developmental divergence of distinct melanocyte subpopulations and may shed light on the pathogenesis of human ocular melanocytosis.<\/jats:p>","DOI":"10.1242\/dmm.050210","type":"journal-article","created":{"date-parts":[[2023,8,14]],"date-time":"2023-08-14T09:33:50Z","timestamp":1692005630000},"source":"Crossref","is-referenced-by-count":2,"title":["Disrupting Hedgehog signaling in melanocytes by SUFU knockout leads to ocular melanocytosis and anterior segment malformation"],"prefix":"10.1242","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-9707-760X","authenticated-orcid":false,"given":"Weizhuo","family":"Wang","sequence":"first","affiliation":[{"name":"State Key Laboratory of Ophthalmology, Optometry and Visual Science, Eye Hospital, Wenzhou Medical University 1 Laboratory of Developmental Cell Biology and Disease , , Wenzhou 325027 , China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Feiyang","family":"Li","sequence":"additional","affiliation":[{"name":"State Key Laboratory of Ophthalmology, Optometry and Visual Science, Eye Hospital, Wenzhou Medical University 1 Laboratory of Developmental Cell Biology and Disease , , Wenzhou 325027 , China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jing","family":"Wang","sequence":"additional","affiliation":[{"name":"State Key Laboratory of Ophthalmology, Optometry and Visual Science, Eye Hospital, Wenzhou Medical University 1 Laboratory of Developmental Cell Biology and Disease , , Wenzhou 325027 , China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Zuimeng","family":"Liu","sequence":"additional","affiliation":[{"name":"State Key Laboratory of Ophthalmology, Optometry and Visual Science, Eye Hospital, Wenzhou Medical University 1 Laboratory of Developmental Cell Biology and Disease , , Wenzhou 325027 , China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Meiyu","family":"Tian","sequence":"additional","affiliation":[{"name":"State Key Laboratory of Ophthalmology, Optometry and Visual Science, Eye Hospital, Wenzhou Medical University 1 Laboratory of Developmental Cell Biology and Disease , , Wenzhou 325027 , China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Zhenhang","family":"Wang","sequence":"additional","affiliation":[{"name":"State Key Laboratory of Ophthalmology, Optometry and Visual Science, Eye Hospital, Wenzhou Medical University 1 Laboratory of Developmental Cell Biology and Disease , , Wenzhou 325027 , China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Huirong","family":"Li","sequence":"additional","affiliation":[{"name":"State Key Laboratory of Ophthalmology, Optometry and Visual Science, Eye Hospital, Wenzhou Medical University 1 Laboratory of Developmental Cell Biology and Disease , , Wenzhou 325027 , China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1678-966X","authenticated-orcid":false,"given":"Jia","family":"Qu","sequence":"additional","affiliation":[{"name":"National Clinical Research Center for Ocular Diseases, Eye Hospital, Wenzhou Medical University 2 , Wenzhou 325027 , China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9910-6942","authenticated-orcid":false,"given":"Yu","family":"Chen","sequence":"additional","affiliation":[{"name":"State Key Laboratory of Ophthalmology, Optometry and Visual Science, Eye Hospital, Wenzhou Medical University 1 Laboratory of Developmental Cell Biology and Disease , , Wenzhou 325027 , China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0705-8099","authenticated-orcid":false,"given":"Ling","family":"Hou","sequence":"additional","affiliation":[{"name":"State Key Laboratory of Ophthalmology, Optometry and Visual Science, Eye Hospital, Wenzhou Medical University 1 Laboratory of Developmental Cell Biology and Disease , , Wenzhou 325027 , China"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","published-online":{"date-parts":[[2023,8,29]]},"reference":[{"key":"2023082911042724200_DMM050210C1","doi-asserted-by":"publisher","first-page":"171","DOI":"10.1111\/j.1755-148X.2010.00679.x","article-title":"Stepping up melanocytes to the challenge of UV exposure","volume":"23","author":"Abdel-Malek","year":"2010","journal-title":"Pigment Cell Melanoma Res."},{"key":"2023082911042724200_DMM050210C2","doi-asserted-by":"publisher","first-page":"291","DOI":"10.1007\/s00417-022-05743-1","article-title":"An update on ophthalmological perspectives in oculodermal melanocytosis (Nevus of Ota)","volume":"261","author":"Abdolrahimzadeh","year":"2023","journal-title":"Graefes Arch. Clin. Exp. Ophthalmol."},{"key":"2023082911042724200_DMM050210C3","doi-asserted-by":"publisher","first-page":"425","DOI":"10.1172\/JCI69404","article-title":"Melanocyte-secreted fibromodulin promotes an angiogenic microenvironment","volume":"124","author":"Adini","year":"2014","journal-title":"J. Clin. Invest."},{"key":"2023082911042724200_DMM050210C4","doi-asserted-by":"publisher","first-page":"1148","DOI":"10.1093\/jnci\/djq257","article-title":"GLI2-mediated melanoma invasion and metastasis","volume":"102","author":"Alexaki","year":"2010","journal-title":"J. Natl. Cancer Inst."},{"key":"2023082911042724200_DMM050210C5","doi-asserted-by":"publisher","first-page":"2511","DOI":"10.1242\/dev.037168","article-title":"Two distinct types of mouse melanocyte: differential signaling requirement for the maintenance of non-cutaneous and dermal versus epidermal melanocytes","volume":"136","author":"Aoki","year":"2009","journal-title":"Development"},{"key":"2023082911042724200_DMM050210C6","doi-asserted-by":"publisher","first-page":"1310","DOI":"10.1002\/ajmg.a.33239","article-title":"Sonic Hedgehog mutations are an uncommon cause of developmental eye anomalies","volume":"152A","author":"Bakrania","year":"2010","journal-title":"Am. J. Med. Genet. A"},{"key":"2023082911042724200_DMM050210C7","doi-asserted-by":"publisher","first-page":"215","DOI":"10.1111\/j.1600-0749.2004.00147.x","article-title":"Spotlight on spotted mice: a review of white spotting mouse mutants and associated human pigmentation disorders","volume":"17","author":"Baxter","year":"2004","journal-title":"Pigment Cell Res."},{"key":"2023082911042724200_DMM050210C8","doi-asserted-by":"publisher","first-page":"226","DOI":"10.1007\/BF00176293","article-title":"Hemi-naevus of Ota: perturbation of neural crest differentiation as a likely mechanism","volume":"230","author":"Benson","year":"1992","journal-title":"Graefes Arch. Clin. Exp. Ophthalmol."},{"key":"2023082911042724200_DMM050210C9","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1080\/15513819709168343","article-title":"Neurocristopathy: its growth and development in 20 years","volume":"17","author":"Bolande","year":"1997","journal-title":"Pediatr. Pathol. Lab. Med."},{"key":"2023082911042724200_DMM050210C10","doi-asserted-by":"publisher","first-page":"2","DOI":"10.1016\/j.ydbio.2011.12.042","article-title":"Development and evolution of the neural crest: an overview","volume":"366","author":"Bronner","year":"2012","journal-title":"Dev. Biol."},{"key":"2023082911042724200_DMM050210C11","doi-asserted-by":"publisher","first-page":"4272","DOI":"10.1038\/ncomms5272","article-title":"Cdon acts as a Hedgehog decoy receptor during proximal-distal patterning of the optic vesicle","volume":"5","author":"Cardozo","year":"2014","journal-title":"Nat. Commun."},{"key":"2023082911042724200_DMM050210C12","doi-asserted-by":"publisher","first-page":"917","DOI":"10.1007\/s00439-018-1918-8","article-title":"The Hedgehog pathway and ocular developmental anomalies","volume":"138","author":"Cavodeassi","year":"2019","journal-title":"Hum. Genet."},{"key":"2023082911042724200_DMM050210C13","doi-asserted-by":"publisher","first-page":"103","DOI":"10.1007\/s11010-016-2680-7","article-title":"The transcription factor TBX2 regulates melanogenesis in melanocytes by repressing Oca2","volume":"415","author":"Chen","year":"2016","journal-title":"Mol. Cell. Biochem."},{"key":"2023082911042724200_DMM050210C14","doi-asserted-by":"publisher","first-page":"dmm035642","DOI":"10.1242\/dmm.035642","article-title":"Photoreceptor degeneration in microphthalmia (Mitf) mice: partial rescue by pigment epithelium-derived factor","volume":"12","author":"Chen","year":"2019","journal-title":"Dis. Model. Mech."},{"key":"2023082911042724200_DMM050210C15","doi-asserted-by":"publisher","first-page":"29","DOI":"10.1016\/S0065-230X(08)00402-8","article-title":"Role and regulation of human tumor suppressor SUFU in Hedgehog signaling","volume":"101","author":"Cheng","year":"2008","journal-title":"Adv. Cancer Res."},{"key":"2023082911042724200_DMM050210C16","doi-asserted-by":"publisher","first-page":"315","DOI":"10.2478\/s11535-013-0141-1","article-title":"Heterogeneity of neural crest-derived melanocytes","volume":"8","author":"Cichorek","year":"2013","journal-title":"Open Life Sci."},{"key":"2023082911042724200_DMM050210C17","doi-asserted-by":"publisher","first-page":"305","DOI":"10.1111\/j.1600-0749.2007.00387.x","article-title":"Flanking genomic region of Tyr::Cre mice, rapid genotyping for homozygous mice","volume":"20","author":"Colombo","year":"2007","journal-title":"Pigment Cell Res."},{"key":"2023082911042724200_DMM050210C18","doi-asserted-by":"publisher","first-page":"4407","DOI":"10.1242\/dev.02021","article-title":"Cardiac and CNS defects in a mouse with targeted disruption of suppressor of fused","volume":"132","author":"Cooper","year":"2005","journal-title":"Development"},{"key":"2023082911042724200_DMM050210C19","doi-asserted-by":"publisher","first-page":"3006","DOI":"10.1016\/j.celrep.2019.05.016","article-title":"GLI2 modulated by SUFU and SPOP induces intestinal stem cell niche signals in development and tumorigenesis","volume":"27","author":"Coquenlorge","year":"2019","journal-title":"Cell Rep."},{"key":"2023082911042724200_DMM050210C20","doi-asserted-by":"publisher","first-page":"10050","DOI":"10.1073\/pnas.97.18.10050","article-title":"Neural crest-directed gene transfer demonstrates Wnt1 role in melanocyte expansion and differentiation during mouse development","volume":"97","author":"Dunn","year":"2000","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2023082911042724200_DMM050210C21","doi-asserted-by":"publisher","first-page":"dev169821","DOI":"10.1242\/dev.169821","article-title":"The diverse neural crest: from embryology to human pathology","volume":"146","author":"Etchevers","year":"2019","journal-title":"Development"},{"key":"2023082911042724200_DMM050210C22","doi-asserted-by":"publisher","first-page":"115","DOI":"10.1016\/j.exer.2014.04.019","article-title":"The ciliary marginal zone (CMZ) in development and regeneration of the vertebrate eye","volume":"123","author":"Fischer","year":"2014","journal-title":"Exp. Eye Res."},{"key":"2023082911042724200_DMM050210C23","doi-asserted-by":"publisher","first-page":"983","DOI":"10.1101\/gad.324657.119","article-title":"MITF-the first 25 years","volume":"33","author":"Goding","year":"2019","journal-title":"Genes Dev."},{"key":"2023082911042724200_DMM050210C24","doi-asserted-by":"publisher","first-page":"1410","DOI":"10.1126\/science.1194472","article-title":"Frequent mutation of BAP1 in metastasizing uveal melanomas","volume":"330","author":"Harbour","year":"2010","journal-title":"Science"},{"key":"2023082911042724200_DMM050210C25","doi-asserted-by":"publisher","first-page":"395","DOI":"10.1016\/0092-8674(93)90429-T","article-title":"Mutations at the mouse microphthalmia locus are associated with defects in a gene encoding a novel basic-helix-loop-helix-zipper protein","volume":"74","author":"Hodgkinson","year":"1993","journal-title":"Cell"},{"key":"2023082911042724200_DMM050210C26","doi-asserted-by":"publisher","first-page":"1163","DOI":"10.1038\/cr.2008.303","article-title":"Transcriptional and signaling regulation in neural crest stem cell-derived melanocyte development: do all roads lead to Mitf?","volume":"18","author":"Hou","year":"2008","journal-title":"Cell Res."},{"key":"2023082911042724200_DMM050210C27","doi-asserted-by":"publisher","first-page":"5379","DOI":"10.1242\/dev.127.24.5379","article-title":"Signaling and transcriptional regulation in the neural crest-derived melanocyte lineage: interactions between KIT and MITF","volume":"127","author":"Hou","year":"2000","journal-title":"Development"},{"key":"2023082911042724200_DMM050210C28","doi-asserted-by":"publisher","first-page":"9081","DOI":"10.1073\/pnas.0603114103","article-title":"Interspecies difference in the regulation of melanocyte development by SOX10 and MITF","volume":"103","author":"Hou","year":"2006","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2023082911042724200_DMM050210C29","doi-asserted-by":"publisher","first-page":"670","DOI":"10.1101\/gad.1902910","article-title":"The output of Hedgehog signaling is controlled by the dynamic association between Suppressor of Fused and the Gli proteins","volume":"24","author":"Humke","year":"2010","journal-title":"Genes Dev."},{"key":"2023082911042724200_DMM050210C30","doi-asserted-by":"publisher","first-page":"966","DOI":"10.1038\/40146","article-title":"Wnt signalling required for expansion of neural crest and CNS progenitors","volume":"389","author":"Ikeya","year":"1997","journal-title":"Nature"},{"key":"2023082911042724200_DMM050210C31","doi-asserted-by":"publisher","first-page":"393","DOI":"10.1038\/nrg2984","article-title":"Mechanisms and functions of Hedgehog signalling across the metazoa","volume":"12","author":"Ingham","year":"2011","journal-title":"Nat. Rev. Genet."},{"key":"2023082911042724200_DMM050210C32","doi-asserted-by":"publisher","first-page":"24","DOI":"10.1038\/s41572-020-0158-0","article-title":"Uveal melanoma","volume":"6","author":"Jager","year":"2020","journal-title":"Nat. Rev. Dis. Primers"},{"key":"2023082911042724200_DMM050210C33","doi-asserted-by":"publisher","first-page":"452","DOI":"10.1016\/j.ydbio.2009.04.009","article-title":"Suppressor of Fused inhibits mammalian Hedgehog signaling in the absence of cilia","volume":"330","author":"Jia","year":"2009","journal-title":"Dev. Biol."},{"key":"2023082911042724200_DMM050210C34","doi-asserted-by":"publisher","first-page":"445","DOI":"10.1038\/s41586-018-0213-0","article-title":"Protection from UV light is an evolutionarily conserved feature of the haematopoietic niche","volume":"558","author":"Kapp","year":"2018","journal-title":"Nature"},{"key":"2023082911042724200_DMM050210C35","doi-asserted-by":"publisher","first-page":"245","DOI":"10.1111\/j.1440-169X.2008.01033.x","article-title":"Wnt signaling in retinal stem cells and regeneration","volume":"50","author":"Kubo","year":"2008","journal-title":"Dev. Growth Differ."},{"key":"2023082911042724200_DMM050210C36","doi-asserted-by":"publisher","first-page":"G1","DOI":"10.1152\/ajpgi.00452.2012","article-title":"Enteric nervous system development: migration, differentiation, and disease","volume":"305","author":"Lake","year":"2013","journal-title":"Am. J. Physiol. Gastrointest. Liver Physiol."},{"key":"2023082911042724200_DMM050210C37","doi-asserted-by":"publisher","first-page":"100846","DOI":"10.1016\/j.preteyeres.2020.100846","article-title":"The cell biology of the retinal pigment epithelium","volume":"78","author":"Lakkaraju","year":"2020","journal-title":"Prog. Retin. Eye Res."},{"key":"2023082911042724200_DMM050210C38","doi-asserted-by":"publisher","first-page":"733","DOI":"10.2741\/1831","article-title":"The WNT\/Beta-catenin pathway in melanoma","volume":"11","author":"Larue","year":"2006","journal-title":"Front. Biosci."},{"key":"2023082911042724200_DMM050210C39","doi-asserted-by":"publisher","first-page":"162","DOI":"10.1016\/S0012-1606(03)00160-X","article-title":"The endothelin receptor-B is required for the migration of neural crest-derived melanocyte and enteric neuron precursors","volume":"259","author":"Lee","year":"2003","journal-title":"Dev. Biol."},{"key":"2023082911042724200_DMM050210C40","doi-asserted-by":"publisher","first-page":"556","DOI":"10.1111\/pcmr.12701","article-title":"Regulation of melanocyte stem cell behavior by the niche microenvironment","volume":"31","author":"Li","year":"2018","journal-title":"Pigment Cell Melanoma Res"},{"key":"2023082911042724200_DMM050210C41","doi-asserted-by":"publisher","first-page":"4507","DOI":"10.1167\/iovs.06-0391","article-title":"Uveal melanocytes do not respond to or express receptors for alpha-melanocyte-stimulating hormone","volume":"47","author":"Li","year":"2006","journal-title":"Invest. Ophthalmol. Vis. Sci."},{"key":"2023082911042724200_DMM050210C42","doi-asserted-by":"publisher","first-page":"173","DOI":"10.1016\/j.exer.2011.01.009","article-title":"Comparative effects of TGF-\u03b22\/Smad2 and TGF-\u03b22\/Smad3 signaling pathways on proliferation, migration, and extracellular matrix production in a human lens cell line","volume":"92","author":"Li","year":"2011","journal-title":"Exp. Eye Res."},{"key":"2023082911042724200_DMM050210C43","doi-asserted-by":"publisher","first-page":"e0128006","DOI":"10.1371\/journal.pone.0128006","article-title":"Suppressor of Fused is required for determining digit number and identity via Gli3\/Fgfs\/Gremlin","volume":"10","author":"Li","year":"2015","journal-title":"PLoS One"},{"key":"2023082911042724200_DMM050210C44","doi-asserted-by":"publisher","first-page":"7272","DOI":"10.1038\/s41598-017-07683-x","article-title":"Epilation induces hair and skin pigmentation through an EDN3\/EDNRB-dependent regenerative response of melanocyte stem cells","volume":"7","author":"Li","year":"2017","journal-title":"Sci. Rep."},{"key":"2023082911042724200_DMM050210C45","doi-asserted-by":"publisher","first-page":"15814","DOI":"10.1074\/jbc.M117.777532","article-title":"Suppressor of Fused restraint of Hedgehog activity level is critical for osteogenic proliferation and differentiation during calvarial bone development","volume":"292","author":"Li","year":"2017","journal-title":"J. Biol. Chem."},{"key":"2023082911042724200_DMM050210C46","doi-asserted-by":"publisher","first-page":"e51698","DOI":"10.7554\/eLife.51698","article-title":"KIT ligand protects against both light-induced and genetic photoreceptor degeneration","volume":"9","author":"Li","year":"2020","journal-title":"Elife"},{"key":"2023082911042724200_DMM050210C47","first-page":"483","article-title":"MAP kinase and beta-catenin signaling in HGF induced RPE migration","volume":"8","author":"Liou","year":"2002","journal-title":"Mol. Vis."},{"key":"2023082911042724200_DMM050210C48","doi-asserted-by":"publisher","first-page":"88","DOI":"10.1038\/emboj.2008.266","article-title":"TGF\u03b2-stimulated Smad1\/5 phosphorylation requires the ALK5 L45 loop and mediates the pro-migratory TGF\u03b2 switch","volume":"28","author":"Liu","year":"2009","journal-title":"EMBO J."},{"key":"2023082911042724200_DMM050210C49","doi-asserted-by":"publisher","first-page":"11681","DOI":"10.18632\/oncotarget.2585","article-title":"Suppressor of fused (Sufu) represses Gli1 transcription and nuclear accumulation, inhibits glioma cell proliferation, invasion and vasculogenic mimicry, improving glioma chemo-sensitivity and prognosis","volume":"5","author":"Liu","year":"2014","journal-title":"Oncotarget"},{"key":"2023082911042724200_DMM050210C50","doi-asserted-by":"publisher","first-page":"1837","DOI":"10.1053\/j.gastro.2015.07.060","article-title":"Identification of GLI mutations in patients with Hirschsprung disease that disrupt enteric nervous system development in mice","volume":"149","author":"Liu","year":"2015","journal-title":"Gastroenterology"},{"key":"2023082911042724200_DMM050210C51","doi-asserted-by":"publisher","first-page":"100766","DOI":"10.1016\/j.preteyeres.2019.06.002","article-title":"The transcription factor MITF in RPE function and dysfunction","volume":"73","author":"Ma","year":"2019","journal-title":"Prog. Retin. Eye Res."},{"key":"2023082911042724200_DMM050210C52","doi-asserted-by":"publisher","first-page":"e0119455","DOI":"10.1371\/journal.pone.0119455","article-title":"T396I mutation of mouse Sufu reduces the stability and activity of Gli3 repressor","volume":"10","author":"Makino","year":"2015","journal-title":"PLoS One"},{"key":"2023082911042724200_DMM050210C53","doi-asserted-by":"publisher","first-page":"e231376","DOI":"10.1136\/bcr-2019-231376","article-title":"Congenital choroidal melanocytoma in a child with ocular melanocytosis","volume":"12","author":"Maniar","year":"2019","journal-title":"BMJ Case Rep."},{"key":"2023082911042724200_DMM050210C54","doi-asserted-by":"publisher","first-page":"3153","DOI":"10.1016\/j.celrep.2016.11.016","article-title":"The ciliary margin zone of the mammalian retina generates retinal ganglion cells","volume":"17","author":"Marcucci","year":"2016","journal-title":"Cell Rep."},{"key":"2023082911042724200_DMM050210C55","doi-asserted-by":"publisher","first-page":"136","DOI":"10.1186\/1471-2350-10-136","article-title":"Reduced transcription of TCOF1 in adult cells of Treacher Collins syndrome patients","volume":"10","author":"Masotti","year":"2009","journal-title":"BMC Med. Genet."},{"key":"2023082911042724200_DMM050210C56","doi-asserted-by":"publisher","first-page":"2118","DOI":"10.1093\/hmg\/ddn110","article-title":"A sensitized mutagenesis screen identifies Gli3 as a modifier of Sox10 neurocristopathy","volume":"17","author":"Matera","year":"2008","journal-title":"Hum. Mol. Genet."},{"key":"2023082911042724200_DMM050210C57","doi-asserted-by":"publisher","first-page":"540","DOI":"10.1167\/iovs.02-0570","article-title":"HGF, MAPK, and a small physiological electric field interact during corneal epithelial cell migration","volume":"44","author":"Mcbain","year":"2003","journal-title":"Invest. Ophthalmol. Vis. Sci."},{"key":"2023082911042724200_DMM050210C58","doi-asserted-by":"publisher","first-page":"40113","DOI":"10.1074\/jbc.M105317200","article-title":"Suppressor of fused negatively regulates beta-catenin signaling","volume":"276","author":"Meng","year":"2001","journal-title":"J. Biol. Chem."},{"key":"2023082911042724200_DMM050210C59","doi-asserted-by":"publisher","first-page":"66","DOI":"10.1002\/dvdy.20299","article-title":"Sonic Hedgehog regulates proliferation of the retinal ciliary marginal zone in posthatch chicks","volume":"233","author":"Moshiri","year":"2005","journal-title":"Dev. Dyn."},{"key":"2023082911042724200_DMM050210C60","doi-asserted-by":"publisher","first-page":"562","DOI":"10.1016\/S0962-8924(02)02405-4","article-title":"Pathways and consequences: Hedgehog signaling in human disease","volume":"12","author":"Mullor","year":"2002","journal-title":"Trends Cell Biol."},{"key":"2023082911042724200_DMM050210C61","doi-asserted-by":"publisher","first-page":"144","DOI":"10.1016\/j.preteyeres.2009.12.002","article-title":"The multifunctional choroid","volume":"29","author":"Nickla","year":"2010","journal-title":"Prog. Retin. Eye Res."},{"key":"2023082911042724200_DMM050210C62","doi-asserted-by":"publisher","first-page":"182","DOI":"10.1634\/theoncologist.9-2-182","article-title":"Recent advances in melanoma biology","volume":"9","author":"Perlis","year":"2004","journal-title":"Oncologist"},{"key":"2023082911042724200_DMM050210C63","doi-asserted-by":"publisher","first-page":"e2107006119","DOI":"10.1073\/pnas.2107006119","article-title":"MITF deficiency accelerates GNAQ-driven uveal melanoma","volume":"119","author":"Phelps","year":"2022","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2023082911042724200_DMM050210C64","doi-asserted-by":"publisher","first-page":"524","DOI":"10.3109\/08820538.2015.1118133","article-title":"An update on oculodermal melanocytosis and rare associated conditions","volume":"32","author":"Plateroti","year":"2017","journal-title":"Semin. Ophthalmol."},{"key":"2023082911042724200_DMM050210C65","doi-asserted-by":"publisher","first-page":"941","DOI":"10.1016\/j.cell.2011.05.004","article-title":"Coordinated activation of Wnt in epithelial and melanocyte stem cells initiates pigmented hair regeneration","volume":"145","author":"Rabbani","year":"2011","journal-title":"Cell"},{"key":"2023082911042724200_DMM050210C66","doi-asserted-by":"publisher","first-page":"24","DOI":"10.1038\/nrn704","article-title":"Hedgehog-Gli signalling and the growth of the brain","volume":"3","author":"Ruiz i Altaba","year":"2002","journal-title":"Nat. Rev. Neurosci."},{"key":"2023082911042724200_DMM050210C67","doi-asserted-by":"publisher","first-page":"160","DOI":"10.1111\/j.1755-148X.2010.00678.x","article-title":"Roles of endothelin signaling in melanocyte development and melanoma","volume":"23","author":"Saldana-Caboverde","year":"2010","journal-title":"Pigment Cell Melanoma Res."},{"key":"2023082911042724200_DMM050210C68","first-page":"756","article-title":"Constitutive mitogen-activated protein kinase activation in melanoma is mediated by both BRAF mutations and autocrine growth factor stimulation","volume":"63","author":"Satyamoorthy","year":"2003","journal-title":"Cancer Res."},{"key":"2023082911042724200_DMM050210C69","doi-asserted-by":"publisher","first-page":"310","DOI":"10.3109\/02713683.2010.542869","article-title":"Intracellular signaling in human iridial fibroblasts and iridial melanocytes in response to prostaglandins, endothelin, isoproterenol, and other pharmacological agents","volume":"36","author":"Sharif","year":"2011","journal-title":"Curr. Eye Res."},{"key":"2023082911042724200_DMM050210C70","doi-asserted-by":"publisher","first-page":"51","DOI":"10.1266\/ggs.17-00058","article-title":"Melanocytes contribute to the vasculature of the choroid","volume":"93","author":"Shibuya","year":"2018","journal-title":"Genes Genet. Syst."},{"key":"2023082911042724200_DMM050210C71","doi-asserted-by":"publisher","first-page":"993","DOI":"10.1001\/jamaophthalmol.2013.129","article-title":"Association of ocular and oculodermal melanocytosis with the rate of uveal melanoma metastasis: analysis of 7872 consecutive eyes","volume":"131","author":"Shields","year":"2013","journal-title":"JAMA Ophthalmol."},{"key":"2023082911042724200_DMM050210C72","doi-asserted-by":"publisher","first-page":"1881","DOI":"10.1016\/j.ophtha.2011.01.040","article-title":"Uveal melanoma: trends in incidence, treatment, and survival","volume":"118","author":"Singh","year":"2011","journal-title":"Ophthalmology"},{"key":"2023082911042724200_DMM050210C73","doi-asserted-by":"publisher","first-page":"172","DOI":"10.1111\/1523-1747.ep12460956","article-title":"Differential expression and activity of melanogenesis-related proteins during induced hair growth in mice","volume":"96","author":"Slominski","year":"1991","journal-title":"J. Invest. Dermatol."},{"key":"2023082911042724200_DMM050210C74","doi-asserted-by":"publisher","first-page":"862","DOI":"10.1111\/1523-1747.ep12382606","article-title":"Melanogenesis during the anagen-catagen-telogen transformation of the murine hair cycle","volume":"102","author":"Slominski","year":"1994","journal-title":"J. Invest. Dermatol."},{"key":"2023082911042724200_DMM050210C75","doi-asserted-by":"publisher","first-page":"1203","DOI":"10.1111\/1523-1747.ep12348479","article-title":"Pharmacological disruption of hair follicle pigmentation by cyclophosphamide as a model for studying the melanocyte response to and recovery from cytotoxic drug damage in situ","volume":"106","author":"Slominski","year":"1996","journal-title":"J. Invest. Dermatol."},{"key":"2023082911042724200_DMM050210C76","doi-asserted-by":"publisher","first-page":"1155","DOI":"10.1152\/physrev.00044.2003","article-title":"Melanin pigmentation in mammalian skin and its hormonal regulation","volume":"84","author":"Slominski","year":"2004","journal-title":"Physiol. Rev."},{"key":"2023082911042724200_DMM050210C77","doi-asserted-by":"publisher","first-page":"C1757","DOI":"10.1152\/ajpcell.00147.2022","article-title":"Neuroendocrine signaling in the skin with a special focus on the epidermal neuropeptides","volume":"323","author":"Slominski","year":"2022","journal-title":"Am. J. Physiol. Cell Physiol."},{"key":"2023082911042724200_DMM050210C78","doi-asserted-by":"publisher","first-page":"842496","DOI":"10.3389\/fonc.2022.842496","article-title":"Melanoma, melanin, and melanogenesis: the Yin and Yang relationship","volume":"12","author":"Slominski","year":"2022","journal-title":"Front. Oncol."},{"key":"2023082911042724200_DMM050210C79","doi-asserted-by":"publisher","first-page":"14","DOI":"10.3390\/jdb5040014","article-title":"The Hedgehog signaling pathway emerges as a pathogenic target","volume":"5","author":"Smelkinson","year":"2017","journal-title":"J. Dev. Biol."},{"key":"2023082911042724200_DMM050210C80","doi-asserted-by":"publisher","first-page":"pe42","DOI":"10.1126\/stke.2982005pe42","article-title":"Checkpoints of melanocyte stem cell development","volume":"2005","author":"Sommer","year":"2005","journal-title":"Sci. STKE"},{"key":"2023082911042724200_DMM050210C81","doi-asserted-by":"publisher","first-page":"5895","DOI":"10.1073\/pnas.0700776104","article-title":"Melanomas require Hedgehog-GLI signaling regulated by interactions between GLI1 and the RAS-MEK\/AKT pathways","volume":"104","author":"Stecca","year":"2007","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2023082911042724200_DMM050210C82","doi-asserted-by":"publisher","first-page":"187","DOI":"10.1016\/j.devcel.2005.12.013","article-title":"Genetic elimination of Suppressor of fused reveals an essential repressor function in the mammalian Hedgehog signaling pathway","volume":"10","author":"Sv\u00e4rd","year":"2006","journal-title":"Dev. Cell"},{"key":"2023082911042724200_DMM050210C83","doi-asserted-by":"publisher","first-page":"599","DOI":"10.1038\/nature07586","article-title":"Frequent somatic mutations of GNAQ in uveal melanoma and blue naevi","volume":"457","author":"Van Raamsdonk","year":"2009","journal-title":"Nature"},{"key":"2023082911042724200_DMM050210C84","doi-asserted-by":"publisher","first-page":"2001","DOI":"10.1242\/dev.052126","article-title":"Suppressor of fused and Spop regulate the stability, processing and function of Gli2 and Gli3 full-length activators but not their repressors","volume":"137","author":"Wang","year":"2010","journal-title":"Development"},{"key":"2023082911042724200_DMM050210C85","doi-asserted-by":"publisher","first-page":"pe11","DOI":"10.1126\/scisignal.2004114","article-title":"A Wnt-er migration: the confusing role of \u03b2-catenin in melanoma metastasis","volume":"6","author":"Webster","year":"2013","journal-title":"Sci. Signal."},{"key":"2023082911042724200_DMM050210C86","doi-asserted-by":"publisher","first-page":"287","DOI":"10.1034\/j.1600-0749.2003.00055.x","article-title":"The role of Kit-ligand in melanocyte development and epidermal homeostasis","volume":"16","author":"Wehrle-Haller","year":"2003","journal-title":"Pigment Cell Res."},{"key":"2023082911042724200_DMM050210C87","doi-asserted-by":"publisher","first-page":"471","DOI":"10.1006\/dbio.2001.0167","article-title":"Analysis of melanocyte precursors in Nf1 mutants reveals that MGF\/KIT signaling promotes directed cell migration independent of its function in cell survival","volume":"232","author":"Wehrle-Haller","year":"2001","journal-title":"Dev. Biol."},{"key":"2023082911042724200_DMM050210C88","doi-asserted-by":"publisher","first-page":"4647","DOI":"10.1038\/s41467-019-12624-5","article-title":"Sufu- and Spop-mediated downregulation of Hedgehog signaling promotes beta cell differentiation through organ-specific niche signals","volume":"10","author":"Yung","year":"2019","journal-title":"Nat. Commun."},{"key":"2023082911042724200_DMM050210C89","doi-asserted-by":"publisher","first-page":"4435","DOI":"10.1242\/dev.129.19.4435","article-title":"BMP signaling is required for development of the ciliary body","volume":"129","author":"Zhao","year":"2002","journal-title":"Development"}],"container-title":["Disease Models &amp; Mechanisms"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.050210\/3102970\/dmm050210.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.050210\/3113941\/dmm050210.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.050210\/3113941\/dmm050210.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,29]],"date-time":"2023-08-29T11:04:55Z","timestamp":1693307095000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.biologists.com\/dmm\/article\/16\/8\/dmm050210\/326569\/Disrupting-Hedgehog-signaling-in-melanocytes-by"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,8,1]]},"references-count":89,"journal-issue":{"issue":"8","published-print":{"date-parts":[[2023,8,1]]}},"URL":"https:\/\/doi.org\/10.1242\/dmm.050210","relation":{},"ISSN":["1754-8403","1754-8411"],"issn-type":[{"value":"1754-8403","type":"print"},{"value":"1754-8411","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,8,1]]},"article-number":"dmm050210"}}