{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,14]],"date-time":"2026-10-14T12:57:45Z","timestamp":1791982665514,"version":"4.4.0"},"reference-count":53,"publisher":"MDPI AG","issue":"4","license":[{"start":{"date-parts":[[2022,9,22]],"date-time":"2022-09-22T00:00:00Z","timestamp":1663804800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"National Institutes of Health","award":["#P20GM109095"],"award-info":[{"award-number":["#P20GM109095"]}]},{"name":"National Institutes of Health","award":["#R01AR47985"],"award-info":[{"award-number":["#R01AR47985"]}]},{"name":"National Institutes of Health","award":["#K02AR48672"],"award-info":[{"award-number":["#K02AR48672"]}]},{"name":"National Institutes of Health","award":["#R15HD059949"],"award-info":[{"award-number":["#R15HD059949"]}]},{"name":"National Institutes of Health","award":["RRID:SCR_019174"],"award-info":[{"award-number":["RRID:SCR_019174"]}]},{"name":"BSU-Biomolecular Research Center","award":["#P20GM109095"],"award-info":[{"award-number":["#P20GM109095"]}]},{"name":"BSU-Biomolecular Research Center","award":["#R01AR47985"],"award-info":[{"award-number":["#R01AR47985"]}]},{"name":"BSU-Biomolecular Research Center","award":["#K02AR48672"],"award-info":[{"award-number":["#K02AR48672"]}]},{"name":"BSU-Biomolecular Research Center","award":["#R15HD059949"],"award-info":[{"award-number":["#R15HD059949"]}]},{"name":"BSU-Biomolecular Research Center","award":["RRID:SCR_019174"],"award-info":[{"award-number":["RRID:SCR_019174"]}]},{"name":"M. J. Murdock Charitable Trust","award":["#P20GM109095"],"award-info":[{"award-number":["#P20GM109095"]}]},{"name":"M. J. Murdock Charitable Trust","award":["#R01AR47985"],"award-info":[{"award-number":["#R01AR47985"]}]},{"name":"M. J. Murdock Charitable Trust","award":["#K02AR48672"],"award-info":[{"award-number":["#K02AR48672"]}]},{"name":"M. J. Murdock Charitable Trust","award":["#R15HD059949"],"award-info":[{"award-number":["#R15HD059949"]}]},{"name":"M. J. Murdock Charitable Trust","award":["RRID:SCR_019174"],"award-info":[{"award-number":["RRID:SCR_019174"]}]},{"name":"Idaho State Board of Education","award":["#P20GM109095"],"award-info":[{"award-number":["#P20GM109095"]}]},{"name":"Idaho State Board of Education","award":["#R01AR47985"],"award-info":[{"award-number":["#R01AR47985"]}]},{"name":"Idaho State Board of Education","award":["#K02AR48672"],"award-info":[{"award-number":["#K02AR48672"]}]},{"name":"Idaho State Board of Education","award":["#R15HD059949"],"award-info":[{"award-number":["#R15HD059949"]}]},{"name":"Idaho State Board of Education","award":["RRID:SCR_019174"],"award-info":[{"award-number":["RRID:SCR_019174"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JDB"],"abstract":"<jats:p>The expression of the col11a1a gene is essential for normal skeletal development, affecting both cartilage and bone. Loss of function mutations have been shown to cause abnormalities in the growth plate of long bones, as well as in craniofacial development. However, the specific effects on Meckel\u2019s cartilage have not been well studied. To further understand the effect of col11a1a gene function, we analyzed the developing jaw in zebrafish using gene knockdown by the injection of an antisense morpholino oligonucleotide using transgenic Tg(sp7:EGFP) and Tg(Fli1a:EGFP) EGFP reporter fish, as well as wildtype AB zebrafish. Our results demonstrate that zebrafish col11a1a knockdown impairs the cellular organization of Meckel\u2019s cartilage in the developing jaw and alters the bone formation that occurs adjacent to the Meckel\u2019s cartilage. These results suggest roles for Col11a1a protein in cartilage intermediates of bone development, the subsequent mineralization of the bony collar of long bones, and that which occurs adjacent to Meckel\u2019s cartilage in the developing jaw.<\/jats:p>","DOI":"10.3390\/jdb10040040","type":"journal-article","created":{"date-parts":[[2022,9,22]],"date-time":"2022-09-22T21:10:05Z","timestamp":1663881005000},"page":"40","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["The Shape of the Jaw\u2014Zebrafish Col11a1a Regulates Meckel\u2019s Cartilage Morphogenesis and Mineralization"],"prefix":"10.3390","volume":"10","author":[{"given":"Jonathon C.","family":"Reeck","sequence":"first","affiliation":[{"name":"Department of Biological Sciences, Biomolecular Research Center, Center of Biomedical Research Excellence in Matrix Biology, Boise State University, Boise, ID 83725, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4850-3569","authenticated-orcid":false,"given":"Julia Thom","family":"Oxford","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences, Biomolecular Research Center, Center of Biomedical Research Excellence in Matrix Biology, Boise State University, Boise, ID 83725, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2022,9,22]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"245","DOI":"10.1006\/dbio.1998.9016","article-title":"The shaping of pharyngeal cartilages during early development of the zebrafish","volume":"203","author":"Kimmel","year":"1998","journal-title":"Dev. Biol."},{"key":"ref_2","doi-asserted-by":"crossref","unstructured":"Kague, E., Gallagher, M., Burke, S., Parsons, M., Franz-Odendaal, T., and Fisher, S. (2012). Skeletogenic Fate of Zebrafish Cranial and Trunk Neural Crest. PLoS ONE, 7.","DOI":"10.1371\/journal.pone.0047394"},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"14","DOI":"10.1016\/j.bone.2015.04.035","article-title":"Bone development","volume":"80","author":"Berendsen","year":"2015","journal-title":"Bone"},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"483","DOI":"10.1242\/dev.120.3.483","article-title":"Segment and cell type lineage restrictions during pharyngeal arch development in the zebrafish embryo","volume":"120","author":"Schilling","year":"1994","journal-title":"Development"},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"235","DOI":"10.1016\/bs.ctdb.2015.07.001","article-title":"Zebrafish Craniofacial Development: A Window into Early Patterning","volume":"115","author":"Mork","year":"2015","journal-title":"Curr. Top. Dev. Biol."},{"key":"ref_6","doi-asserted-by":"crossref","unstructured":"Eames, B., DeLaurier, A., Ullmann, B., Huycke, T., Nichols, J., Dowd, J., McFadden, M., Sasaki, M., and Kimmel, C. (2013). FishFace: Interactive atlas of zebrafish craniofacial development at cellular resolution. BMC Dev. Biol., 13.","DOI":"10.1186\/1471-213X-13-23"},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"46","DOI":"10.1097\/MOP.0b013e32832185c5","article-title":"Genetic and orthopedic aspects of collagen disorders","volume":"21","author":"Carter","year":"2009","journal-title":"Curr. Opin. Pediatr."},{"key":"ref_8","first-page":"a004978","article-title":"The collagen family","volume":"3","year":"2011","journal-title":"Cold Spring Harb. Perspect. Biol."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"1955","DOI":"10.1242\/jcs.03453","article-title":"Collagens at a glance","volume":"120","author":"Kadler","year":"2007","journal-title":"J. Cell Sci."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"509","DOI":"10.1002\/jor.1100050406","article-title":"Chondrons in cartilage: Ultrastructural analysis of the pericellular microenvironment in adult human articular cartilages","volume":"5","author":"Poole","year":"1987","journal-title":"J. Orthop. Res."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"30","DOI":"10.1186\/ar380","article-title":"Collagen of articular cartilage","volume":"4","author":"Eyre","year":"2002","journal-title":"Arthritis Res."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"708","DOI":"10.1016\/j.ajhg.2010.10.009","article-title":"Fibrochondrogenesis results from mutations in the COL11A1 type XI collagen gene","volume":"87","author":"Tompson","year":"2010","journal-title":"Am. J. Hum. Genet."},{"key":"ref_13","doi-asserted-by":"crossref","unstructured":"Vijzelaar, R., Waller, S., Errami, A., Donaldson, A., Lourenco, T., Rodrigues, M., McConnell, V., Fincham, G., Snead, M., and Richards, A. (2013). Deletions within COL11A1 in Type 2 stickler syndrome detected by multiplex ligation-dependent probe amplification (MLPA). BMC Med. Genet., 14.","DOI":"10.1186\/1471-2350-14-48"},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"230","DOI":"10.1016\/j.ymgme.2014.09.001","article-title":"Novel pathogenic COL11A1\/COL11A2 variants in Stickler syndrome detected by targeted NGS and exome sequencing","volume":"113","author":"Acke","year":"2014","journal-title":"Mol. Genet. Metab."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"265","DOI":"10.1002\/ajmg.1320190209","article-title":"Fibrochondrogenesis: Lethal, autosomal recessive chondrodysplasia with distinctive cartilage histopathology","volume":"19","author":"Whitley","year":"1984","journal-title":"Am. J. Med. Genet."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"147","DOI":"10.1111\/j.1399-0004.2011.01734.x","article-title":"Clinical and molecular analysis of UAE fibrochondrogenesis patients expands the phenotype and reveals two COL11A1 homozygous null mutations","volume":"82","author":"Akawi","year":"2012","journal-title":"Clin. Genet."},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"2607","DOI":"10.1002\/ajmg.a.36688","article-title":"A novel dominant COL11A1 mutation resulting in a severe skeletal dysplasia","volume":"164A","author":"Hufnagel","year":"2014","journal-title":"Am. J. Med. Genet. A"},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"39507","DOI":"10.1074\/jbc.M608551200","article-title":"Isoform-specific heparan sulfate binding within the amino-terminal noncollagenous domain of collagen alpha1(XI)","volume":"281","author":"Warner","year":"2006","journal-title":"J. Biol. Chem."},{"key":"ref_19","first-page":"43","article-title":"Predicted structure and binding motifs of collagen \u03b11(XI)","volume":"1","author":"McDougal","year":"2011","journal-title":"GSTF Int. J. Bioinform. Biotechnol."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"4660","DOI":"10.1002\/pmic.201100058","article-title":"Proteomic analysis of Col11a1-associated protein complexes","volume":"11","author":"Brown","year":"2011","journal-title":"Proteomics"},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"71","DOI":"10.1016\/j.cyto.2014.11.007","article-title":"Oncostatin M binds to extracellular matrix in a bioactive conformation: Implications for inflammation and metastasis","volume":"72","author":"Ryan","year":"2015","journal-title":"Cytokine"},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"597","DOI":"10.1016\/j.matbio.2007.06.007","article-title":"Collagen XI chain misassembly in cartilage of the chondrodysplasia (cho) mouse","volume":"26","author":"Fernandes","year":"2007","journal-title":"Matrix Biol."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"423","DOI":"10.1016\/0092-8674(95)90492-1","article-title":"A fibrillar collagen gene, Col11a1, is essential for skeletal morphogenesis","volume":"80","author":"Li","year":"1995","journal-title":"Cell"},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"315","DOI":"10.1016\/j.gep.2010.07.002","article-title":"The expression patterns of minor fibrillar collagens during development in zebrafish","volume":"10","author":"Fang","year":"2010","journal-title":"Gene Expr. Patterns"},{"key":"ref_25","doi-asserted-by":"crossref","unstructured":"Hardy, M.J., Reeck, J.C., Fang, M., Adams, J.S., and Oxford, J.T. (2020). Col11a1a Expression is required for zebrafish development. J. Dev. Biol., 8.","DOI":"10.3390\/jdb8030016"},{"key":"ref_26","doi-asserted-by":"crossref","unstructured":"Reeck, J.C., Hardy, M.J., Pu, X., Keller-Peck, C., and Oxford, J.T. (2021). Authentication of a novel antibody to zebrafish collagen type XI alpha 1 chain (Col11a1a). BMC Res. Notes, 14.","DOI":"10.1186\/s13104-021-05770-x"},{"key":"ref_27","doi-asserted-by":"crossref","first-page":"505","DOI":"10.1002\/dvg.20639","article-title":"Zebrafish sp7:EGFP: A transgenic for studying otic vesicle formation, skeletogenesis, and bone regeneration","volume":"48","author":"DeLaurier","year":"2010","journal-title":"Genesis"},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"307","DOI":"10.1006\/dbio.2002.0711","article-title":"In Vivo Imaging of Embryonic Vascular Development Using Transgenic Zebrafish","volume":"248","author":"Lawson","year":"2002","journal-title":"Dev. Biol."},{"key":"ref_29","unstructured":"Westerfield, M. (2007). The Zebrafish Book: A Guide for the Laboratory Use of Zebrafish (Brachydanio Rerio), University of Oregon Press. [5th ed.]."},{"key":"ref_30","doi-asserted-by":"crossref","unstructured":"Kimmel, C.B., DeLaurier, A., Ullmann, B., Dowd, J., and McFadden, M. (2010). Modes of developmental outgrowth and shaping of a craniofacial bone in zebrafish. PLoS ONE, 5.","DOI":"10.1371\/journal.pone.0009475"},{"key":"ref_31","first-page":"465","article-title":"Knockdown of Collagen 11a1 decreases zebrafish lens and optic cup diameter during early development","volume":"54","author":"Yonkers","year":"2013","journal-title":"J. Investig. Ophthalmol. Vis. Sci."},{"key":"ref_32","doi-asserted-by":"crossref","first-page":"345","DOI":"10.1242\/dev.123.1.345","article-title":"Jaw and branchial arch mutants in zebrafish II: Anterior arches and cartilage differentiation","volume":"123","author":"Piotrowski","year":"1996","journal-title":"Development"},{"key":"ref_33","doi-asserted-by":"crossref","first-page":"329","DOI":"10.1242\/dev.123.1.329","article-title":"Jaw and branchial arch mutants in zebrafish I: Branchial arches","volume":"123","author":"Schilling","year":"1996","journal-title":"Development"},{"key":"ref_34","doi-asserted-by":"crossref","unstructured":"Eames, B., Amores, A., Yan, Y., and Postlethwait, J.H. (2012). Evolution of the osteoblast: Skeletogenesis in gar and zebrafish. BMC Evol. Biol., 12.","DOI":"10.1186\/1471-2148-12-27"},{"key":"ref_35","doi-asserted-by":"crossref","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"},{"key":"ref_36","doi-asserted-by":"crossref","first-page":"394","DOI":"10.1007\/s11926-013-0394-3","article-title":"The Collagenopathies: Review of Clinical Phenotypes and Molecular Correlations","volume":"16","author":"Jobling","year":"2014","journal-title":"Curr. Rheumatol. Rep."},{"key":"ref_37","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1016\/j.mrrev.2016.02.003","article-title":"Molecular genetics of the COL2A1-related disorders","volume":"768","author":"Deng","year":"2016","journal-title":"Mutat. Res. Mutat. Res."},{"key":"ref_38","doi-asserted-by":"crossref","first-page":"332","DOI":"10.1038\/nature01657","article-title":"Developmental regulation of the growth plate","volume":"423","author":"Kronenberg","year":"2003","journal-title":"Nature"},{"key":"ref_39","unstructured":"Meckel, J.F. (1820). Handbuch der Menschlichem Anatomie, Buchhandlung des Hallischen waisenhauses. Lehre und Geschichte des Foetud."},{"key":"ref_40","doi-asserted-by":"crossref","first-page":"2509","DOI":"10.1002\/art.11233","article-title":"Osteoarthritis-like changes and decreased mechanical function of articular cartilage in the joints of mice with the chondrodysplasia gene (cho)","volume":"48","author":"Xu","year":"2003","journal-title":"Arthritis Rheum."},{"key":"ref_41","doi-asserted-by":"crossref","first-page":"18865","DOI":"10.1074\/jbc.270.32.18865","article-title":"Structural analysis of cross-linking domains in cartilage type XI collagen. Insights on polymeric assembly","volume":"270","author":"Wu","year":"1995","journal-title":"J. Biol. Chem."},{"key":"ref_42","doi-asserted-by":"crossref","first-page":"490","DOI":"10.1016\/j.matbio.2009.07.004","article-title":"Craniofacial cartilage morphogenesis requires zebrafish col11a1 activity","volume":"28","author":"Baas","year":"2009","journal-title":"Matrix Biol."},{"key":"ref_43","doi-asserted-by":"crossref","first-page":"158","DOI":"10.3390\/jdb3040158","article-title":"Col11a1 Regulates Bone Microarchitecture during Embryonic Development","volume":"3","author":"Hafez","year":"2015","journal-title":"J. Dev. Biol."},{"key":"ref_44","doi-asserted-by":"crossref","first-page":"1671","DOI":"10.1242\/dev.127.8.1671","article-title":"Fate of the mammalian cranial neural crest during tooth and mandibular morphogenesis","volume":"127","author":"Chai","year":"2000","journal-title":"Development"},{"key":"ref_45","doi-asserted-by":"crossref","first-page":"55","DOI":"10.1111\/j.1469-8749.1966.tb08273.x","article-title":"The first arch syndrome","volume":"8","author":"McKenzie","year":"1966","journal-title":"Dev. Med. Child Neurol."},{"key":"ref_46","doi-asserted-by":"crossref","first-page":"92","DOI":"10.1016\/j.modgep.2007.10.001","article-title":"Expression of marker genes during otolith development in medaka","volume":"8","author":"Nemoto","year":"2008","journal-title":"Gene Expr. Patterns"},{"key":"ref_47","doi-asserted-by":"crossref","first-page":"84","DOI":"10.1186\/1750-1172-7-84","article-title":"Hearing impairment in Stickler syndrome: A systematic review","volume":"7","author":"Acke","year":"2012","journal-title":"Orphanet J. Rare Dis."},{"key":"ref_48","first-page":"1096","article-title":"Fibrochondrogenesis","volume":"35","year":"1978","journal-title":"Arch. Fr. P\u00e9diatr."},{"key":"ref_49","doi-asserted-by":"crossref","first-page":"418","DOI":"10.1016\/j.ejmg.2012.03.006","article-title":"Mosaicism in Stickler syndrome","volume":"55","author":"Stevenson","year":"2012","journal-title":"Eur. J. Med. Genet."},{"key":"ref_50","doi-asserted-by":"crossref","first-page":"2465","DOI":"10.1101\/gad.277003","article-title":"Beta1 integrins regulate chondrocyte rotation, G1 progression, and cytokinesis","volume":"17","author":"Aszodi","year":"2003","journal-title":"Genes Dev."},{"key":"ref_51","doi-asserted-by":"crossref","first-page":"428","DOI":"10.1016\/j.bbrc.2012.03.012","article-title":"Androgen receptor is up-regulated by a BLT2-linked pathway to contribute to prostate cancer progression","volume":"420","author":"Lee","year":"2012","journal-title":"Biochem. Biophys. Res. Commun."},{"key":"ref_52","doi-asserted-by":"crossref","first-page":"23748","DOI":"10.18632\/oncotarget.4250","article-title":"COL11A1 confers chemoresistance on ovarian cancer cells through the activation of Akt\/c\/EBP\u03b2 pathway and PDK1 stabilization","volume":"6","author":"Wu","year":"2015","journal-title":"Oncotarget"},{"key":"ref_53","doi-asserted-by":"crossref","first-page":"314","DOI":"10.1016\/j.joca.2003.12.002","article-title":"A type XI collagen mutation leads to increased degradation of type II collagen in articular cartilage","volume":"12","author":"Rodriguez","year":"2004","journal-title":"OsteoArthritis Cart"}],"container-title":["Journal of Developmental Biology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2221-3759\/10\/4\/40\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,11]],"date-time":"2025-10-11T00:37:24Z","timestamp":1760143044000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2221-3759\/10\/4\/40"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,9,22]]},"references-count":53,"journal-issue":{"issue":"4","published-online":{"date-parts":[[2022,12]]}},"alternative-id":["jdb10040040"],"URL":"https:\/\/doi.org\/10.3390\/jdb10040040","relation":{},"ISSN":["2221-3759"],"issn-type":[{"value":"2221-3759","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,9,22]]}}}