{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,21]],"date-time":"2026-09-21T10:43:18Z","timestamp":1789987398590,"version":"4.0.1"},"reference-count":69,"publisher":"National Academy of Sciences","issue":"16","license":[{"start":{"date-parts":[[2021,10,15]],"date-time":"2021-10-15T00:00:00Z","timestamp":1634256000000},"content-version":"vor","delay-in-days":183,"URL":"https:\/\/www.pnas.org\/site\/aboutpnas\/licenses.xhtml"}],"funder":[{"DOI":"10.13039\/100006955","name":"Office of Extramural Research, National Institutes of Health","doi-asserted-by":"publisher","award":["R21EY030188"],"award-info":[{"award-number":["R21EY030188"]}],"id":[{"id":"10.13039\/100006955","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["www.pnas.org"],"crossmark-restriction":true},"short-container-title":["Proc. Natl. Acad. Sci. U.S.A."],"published-print":{"date-parts":[[2021,4,20]]},"abstract":"<jats:title>Significance<\/jats:title>\n                  <jats:p>\n                    Mutations in complement factor H (CFH) binding sites for heparan sulfate (HS) increase risk for age-related macular degeneration (AMD), but the roles of CFH and HS in disease pathogenesis remain unclear. Here, we find that a CFH homolog, HS modification, and the HS proteoglycan syndecan\/SDN-1 have roles in maintaining cilia compartment boundaries in the sensory neurons of aging\n                    <jats:italic>C. elegans<\/jats:italic>\n                    adults. The role of CFH in cilia compartment boundaries is conserved in vertebrate photoreceptors, suggesting that structural defects in photoreceptor cilia make a contribution to AMD progression in patients with CFH mutations that has not been appreciated previously.\n                  <\/jats:p>","DOI":"10.1073\/pnas.2016698118","type":"journal-article","created":{"date-parts":[[2021,4,15]],"date-time":"2021-04-15T15:49:52Z","timestamp":1618501792000},"update-policy":"https:\/\/doi.org\/10.1073\/pnas.cm10313","source":"Crossref","is-referenced-by-count":10,"title":["A complement factor H homolog, heparan sulfation, and syndecan maintain inversin compartment boundaries in\n                    <i>C. elegans<\/i>\n                    cilia"],"prefix":"10.1073","volume":"118","author":[{"given":"Natalie","family":"Acker","sequence":"first","affiliation":[{"name":"Center for Biomedical Engineering and Technology, University of Maryland School of Medicine, University of Maryland, Baltimore, MD 21201;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3517-5973","authenticated-orcid":false,"given":"Harold","family":"Smith","sequence":"additional","affiliation":[{"name":"Laboratory of Biochemistry and Genetics, National Institute of Diabetes, Digestive, and Kidney Diseases, NIH, Bethesda, MD 20892;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Claire","family":"Devine","sequence":"additional","affiliation":[{"name":"Center for Biomedical Engineering and Technology, University of Maryland School of Medicine, University of Maryland, Baltimore, MD 21201;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6698-4810","authenticated-orcid":false,"given":"Sharon L.","family":"Oltjen","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology and Vision Science, Vitreoretinal Research Laboratory, University of California, Davis, CA 95616;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3760-9317","authenticated-orcid":false,"given":"Sofia","family":"Tsiropoulou","sequence":"additional","affiliation":[{"name":"School of Biomolecular and Biomedical Research, UCD Conway Institute, University College Dublin, Belfield, Dublin 4, Ireland;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0275-8088","authenticated-orcid":false,"given":"Zeljka","family":"Smit-McBride","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology and Vision Science, Vitreoretinal Research Laboratory, University of California, Davis, CA 95616;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2414-5277","authenticated-orcid":false,"given":"Karen","family":"Lange","sequence":"additional","affiliation":[{"name":"School of Biomolecular and Biomedical Research, UCD Conway Institute, University College Dublin, Belfield, Dublin 4, Ireland;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1598-2695","authenticated-orcid":false,"given":"Oliver E.","family":"Blacque","sequence":"additional","affiliation":[{"name":"School of Biomolecular and Biomedical Research, UCD Conway Institute, University College Dublin, Belfield, Dublin 4, Ireland;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3689-3117","authenticated-orcid":false,"given":"Joanne A.","family":"Matsubara","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology and Visual Sciences, The University of British Columbia, Vancouver, BC V5Z 3N9, Canada;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5550-0286","authenticated-orcid":false,"given":"Andrew","family":"Gordus","sequence":"additional","affiliation":[{"name":"Department of Biology, The Johns Hopkins University, Baltimore, MD 21211;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8599-2031","authenticated-orcid":false,"given":"Andy","family":"Golden","sequence":"additional","affiliation":[{"name":"Laboratory of Biochemistry and Genetics, National Institute of Diabetes, Digestive, and Kidney Diseases, NIH, Bethesda, MD 20892;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2576-3843","authenticated-orcid":false,"given":"Bruce E.","family":"Vogel","sequence":"additional","affiliation":[{"name":"Center for Biomedical Engineering and Technology, University of Maryland School of Medicine, University of Maryland, Baltimore, MD 21201;"},{"name":"Department of Physiology, University of Maryland School of Medicine, University of Maryland, Baltimore, MD 21201"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"341","published-online":{"date-parts":[[2021,4,15]]},"reference":[{"key":"e_1_3_4_1_2","doi-asserted-by":"publisher","DOI":"10.1126\/science.1110189"},{"key":"e_1_3_4_2_2","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0501536102"},{"key":"e_1_3_4_3_2","doi-asserted-by":"publisher","DOI":"10.1126\/science.1110359"},{"key":"e_1_3_4_4_2","doi-asserted-by":"publisher","DOI":"10.1126\/science.1109557"},{"key":"e_1_3_4_5_2","doi-asserted-by":"publisher","DOI":"10.1086\/431426"},{"key":"e_1_3_4_6_2","doi-asserted-by":"publisher","DOI":"10.1038\/ng.976"},{"key":"e_1_3_4_7_2","doi-asserted-by":"publisher","DOI":"10.1146\/annurev-genom-090413-025610"},{"key":"e_1_3_4_8_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.preteyeres.2017.09.001"},{"key":"e_1_3_4_9_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM200104053441406"},{"key":"e_1_3_4_10_2","doi-asserted-by":"publisher","DOI":"10.4049\/jimmunol.1201751"},{"key":"e_1_3_4_11_2","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.biochem.68.1.729"},{"key":"e_1_3_4_12_2","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.cellbio.22.010605.093433"},{"key":"e_1_3_4_13_2","doi-asserted-by":"publisher","DOI":"10.1016\/S1937-6448(09)76003-4"},{"key":"e_1_3_4_14_2","doi-asserted-by":"publisher","DOI":"10.1242\/dev.098178"},{"key":"e_1_3_4_15_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0962-8924(00)01897-3"},{"key":"e_1_3_4_16_2","doi-asserted-by":"publisher","DOI":"10.1159\/000356513"},{"key":"e_1_3_4_17_2","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1424391112"},{"key":"e_1_3_4_18_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.immuni.2017.01.006"},{"key":"e_1_3_4_19_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.ophtha.2018.10.037"},{"key":"e_1_3_4_20_2","doi-asserted-by":"publisher","DOI":"10.1006\/geno.2002.6815"},{"key":"e_1_3_4_21_2","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddu014"},{"key":"e_1_3_4_22_2","doi-asserted-by":"publisher","DOI":"10.1038\/nrm.2017.60"},{"key":"e_1_3_4_23_2","doi-asserted-by":"publisher","DOI":"10.1038\/ng1217"},{"key":"e_1_3_4_24_2","doi-asserted-by":"publisher","DOI":"10.1242\/jcs.037408"},{"key":"e_1_3_4_25_2","first-page":"2592","article-title":"Ciliogenesis in Caenorhabditis elegans requires genetic interactions between ciliary middle segment localized NPHP-2 (inversin) and transition zone-associated proteins","volume":"125","author":"Warburton-Pitt S. R.","year":"2012","unstructured":"S. R. Warburton-Pitt ., Ciliogenesis in Caenorhabditis elegans requires genetic interactions between ciliary middle segment localized NPHP-2 (inversin) and transition zone-associated proteins. J. Cell Sci. 125, 2592\u20132603 (2012).","journal-title":"J. Cell Sci."},{"key":"e_1_3_4_26_2","doi-asserted-by":"publisher","DOI":"10.1038\/2450"},{"key":"e_1_3_4_27_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2011.04.019"},{"key":"e_1_3_4_28_2","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.E19-09-0499"},{"key":"e_1_3_4_29_2","doi-asserted-by":"publisher","DOI":"10.1007\/PL00013152"},{"key":"e_1_3_4_30_2","doi-asserted-by":"publisher","DOI":"10.1534\/genetics.118.301307"},{"key":"e_1_3_4_31_2","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pbio.0050237"},{"key":"e_1_3_4_32_2","doi-asserted-by":"publisher","DOI":"10.1534\/genetics.115.179382"},{"key":"e_1_3_4_33_2","doi-asserted-by":"publisher","DOI":"10.1242\/jcs.028621"},{"key":"e_1_3_4_34_2","first-page":"601","article-title":"The Caenorhabditis elegans gene T23G5.5 encodes an antidepressant- and cocaine-sensitive dopamine transporter","volume":"54","author":"Jayanthi L. D.","year":"1998","unstructured":"L. D. Jayanthi ., The Caenorhabditis elegans gene T23G5.5 encodes an antidepressant- and cocaine-sensitive dopamine transporter. Mol. Pharmacol. 54, 601\u2013609 (1998).","journal-title":"Mol. Pharmacol."},{"key":"e_1_3_4_35_2","doi-asserted-by":"publisher","DOI":"10.1242\/jcs.127274"},{"key":"e_1_3_4_36_2","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.1004866"},{"key":"e_1_3_4_37_2","doi-asserted-by":"publisher","DOI":"10.1038\/nature04538"},{"key":"e_1_3_4_38_2","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.1003977"},{"key":"e_1_3_4_39_2","doi-asserted-by":"publisher","DOI":"10.1534\/g3.112.003830"},{"key":"e_1_3_4_40_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0896-6273(00)81199-X"},{"key":"e_1_3_4_41_2","doi-asserted-by":"publisher","DOI":"10.1038\/srep26297"},{"key":"e_1_3_4_42_2","doi-asserted-by":"publisher","DOI":"10.1534\/genetics.117.300179"},{"key":"e_1_3_4_43_2","doi-asserted-by":"publisher","DOI":"10.1534\/g3.112.005199"},{"key":"e_1_3_4_44_2","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M611107200"},{"key":"e_1_3_4_45_2","doi-asserted-by":"publisher","DOI":"10.1242\/dev.02042"},{"key":"e_1_3_4_46_2","doi-asserted-by":"publisher","DOI":"10.1167\/iovs.05-1655"},{"key":"e_1_3_4_47_2","first-page":"110","article-title":"Localization of complement factor H gene expression and protein distribution in the mouse outer retina","volume":"21","author":"Smit-McBride Z.","year":"2015","unstructured":"Z. Smit-McBride ., Localization of complement factor H gene expression and protein distribution in the mouse outer retina. Mol. Vis. 21, 110\u2013123 (2015).","journal-title":"Mol. Vis."},{"key":"e_1_3_4_48_2","doi-asserted-by":"publisher","DOI":"10.15252\/embj.201593003"},{"key":"e_1_3_4_49_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.kint.2017.11.016"},{"key":"e_1_3_4_50_2","first-page":"264","article-title":"CFH Y402H polymorphism is associated with elevated vitreal GM-CSF and choroidal macrophages in the postmortem human eye","volume":"21","author":"Wang J. C.","year":"2015","unstructured":"J. C. Wang ., CFH Y402H polymorphism is associated with elevated vitreal GM-CSF and choroidal macrophages in the postmortem human eye. Mol. Vis. 21, 264\u2013272 (2015).","journal-title":"Mol. Vis."},{"key":"e_1_3_4_51_2","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/5.8.1171"},{"key":"e_1_3_4_52_2","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/11.16.1899"},{"key":"e_1_3_4_53_2","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M114.611921"},{"key":"e_1_3_4_54_2","doi-asserted-by":"publisher","DOI":"10.1007\/978-94-017-8881-6_3"},{"key":"e_1_3_4_55_2","doi-asserted-by":"publisher","DOI":"10.1038\/nature08430"},{"key":"e_1_3_4_56_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0021-9258(17)31481-3"},{"key":"e_1_3_4_57_2","doi-asserted-by":"publisher","DOI":"10.1534\/genetics.118.300837"},{"key":"e_1_3_4_58_2","doi-asserted-by":"publisher","DOI":"10.1242\/dev.096933"},{"key":"e_1_3_4_59_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.exer.2015.11.007"},{"key":"e_1_3_4_60_2","doi-asserted-by":"publisher","DOI":"10.1093\/ndt\/gfl088"},{"key":"e_1_3_4_61_2","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.2000339117"},{"key":"e_1_3_4_62_2","doi-asserted-by":"publisher","DOI":"10.1080\/13816810.2018.1525753"},{"key":"e_1_3_4_63_2","doi-asserted-by":"publisher","DOI":"10.1534\/genetics.114.169730"},{"key":"e_1_3_4_64_2","doi-asserted-by":"publisher","DOI":"10.1038\/nmeth.2413"},{"key":"e_1_3_4_65_2","doi-asserted-by":"crossref","first-page":"e0174446","DOI":"10.1371\/journal.pone.0174446","article-title":"Evaluating alignment and variant-calling software for mutation identification in C. elegans by whole-genome sequencing","volume":"12","author":"Smith H. E.","year":"2017","unstructured":"H. E. Smith, S. Yun, Evaluating alignment and variant-calling software for mutation identification in C. elegans by whole-genome sequencing. PLoS One 12, e0174446 (2017).","journal-title":"PLoS One"},{"key":"e_1_3_4_66_2","doi-asserted-by":"publisher","DOI":"10.1093\/bioinformatics\/btp352"},{"key":"e_1_3_4_67_2","unstructured":"E. Garrison G. Marth Haplotype-based variant detection from short-read sequencing. arXiv [Preprint] https:\/\/arxiv.org\/abs\/1207.3907 (20 July 2012)."},{"key":"e_1_3_4_68_2","doi-asserted-by":"publisher","DOI":"10.1093\/nar\/gkq603"},{"key":"e_1_3_4_69_2","doi-asserted-by":"publisher","DOI":"10.1016\/bs.mcb.2015.01.010"}],"container-title":["Proceedings of the National Academy of Sciences"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/www.pnas.org\/syndication\/doi\/10.1073\/pnas.2016698118","content-type":"unspecified","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/pnas.org\/doi\/pdf\/10.1073\/pnas.2016698118","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,4,13]],"date-time":"2022-04-13T06:02:14Z","timestamp":1649829734000},"score":1,"resource":{"primary":{"URL":"https:\/\/pnas.org\/doi\/full\/10.1073\/pnas.2016698118"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,4,15]]},"references-count":69,"journal-issue":{"issue":"16","published-print":{"date-parts":[[2021,4,20]]}},"alternative-id":["10.1073\/pnas.2016698118"],"URL":"https:\/\/doi.org\/10.1073\/pnas.2016698118","relation":{},"ISSN":["0027-8424","1091-6490"],"issn-type":[{"value":"0027-8424","type":"print"},{"value":"1091-6490","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,4,15]]},"assertion":[{"value":"2021-04-15","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e2016698118"}}