{"id":"https://openalex.org/W4407946372","doi":"https://doi.org/10.1083/jcb.202403022","title":"Centriole structural integrity defects are a crucial feature of hydrolethalus syndrome","display_name":"Centriole structural integrity defects are a crucial feature of hydrolethalus syndrome","publication_year":2025,"publication_date":"2025-01-22","ids":{"openalex":"https://openalex.org/W4407946372","doi":"https://doi.org/10.1083/jcb.202403022","pmid":"https://pubmed.ncbi.nlm.nih.gov/38496445","pmcid":"https://www.ncbi.nlm.nih.gov/pmc/articles/10942441"},"language":"en","primary_location":{"id":"doi:10.1083/jcb.202403022","doi":"https://doi.org/10.1083/jcb.202403022","is_oa":false,"landing_page_url":"https://doi.org/10.1083/jcb.202403022","pdf_url":null,"source":{"id":"https://openalex.org/S96410829","display_name":"The Journal of Cell Biology","issn_l":"0021-9525","issn":["0021-9525","1540-8140"],"is_oa":false,"is_in_doaj":false,"is_core":true,"listed_in":["anvur-scientific","cwts-core","dongbi-a","doyens","jufo-3","ki-jl-2","medline","norway-2","poland-140","russia-white-list-1"],"host_organization":"https://openalex.org/P4310315676","host_organization_name":"Rockefeller University Press","host_organization_lineage":["https://openalex.org/P4310315676"],"host_organization_lineage_names":["Rockefeller University Press"],"type":"journal"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Journal of Cell Biology","raw_type":"journal-article"},"volume":null,"type":"article","is_traditional":true,"indexed_in":["crossref","pubmed"],"open_access":{"is_oa":true,"oa_status":"green","oa_url":"https://www.biorxiv.org/content/biorxiv/early/2024/03/06/2024.03.06.583733.full.pdf","any_repository_has_fulltext":true},"authorships":[{"author_position":"first","author":{"id":"https://openalex.org/A5037731819","display_name":"Ana Curinha","orcid":"https://orcid.org/0009-0000-5745-1601","observed_orcids":["https://orcid.org/0009-0000-5745-1601"]},"institutions":[{"id":"https://openalex.org/I145311948","display_name":"Johns Hopkins University","ror":"https://ror.org/00za53h95","country_code":"US","type":"education","lineage":["https://openalex.org/I145311948"]},{"id":"https://openalex.org/I2799853436","display_name":"Johns Hopkins Medicine","ror":"https://ror.org/037zgn354","country_code":"US","type":"healthcare","lineage":["https://openalex.org/I2799853436"]}],"countries":["US"],"is_corresponding":true,"raw_author_name":"Ana Curinha","raw_affiliation_strings":["Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","Department of Molecular Biology and Genetics, https://ror.org/00za53h95Johns Hopkins University School of Medicine, Baltimore, MD, USA"],"raw_orcid":"https://orcid.org/0009-0000-5745-1601","affiliations":[{"raw_affiliation_string":"Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","institution_ids":["https://openalex.org/I145311948"]},{"raw_affiliation_string":"Department of Molecular Biology and Genetics, https://ror.org/00za53h95Johns Hopkins University School of Medicine, Baltimore, MD, USA","institution_ids":["https://openalex.org/I145311948","https://openalex.org/I2799853436"]}]},{"author_position":"middle","author":{"id":null,"display_name":"Zhaoyu Huang","orcid":"https://orcid.org/0009-0004-9247-4349","observed_orcids":["https://orcid.org/0009-0004-9247-4349"]},"institutions":[{"id":"https://openalex.org/I145311948","display_name":"Johns Hopkins University","ror":"https://ror.org/00za53h95","country_code":"US","type":"education","lineage":["https://openalex.org/I145311948"]},{"id":"https://openalex.org/I2799853436","display_name":"Johns Hopkins Medicine","ror":"https://ror.org/037zgn354","country_code":"US","type":"healthcare","lineage":["https://openalex.org/I2799853436"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Zhaoyu Huang","raw_affiliation_strings":["Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","Department of Molecular Biology and Genetics, https://ror.org/00za53h95Johns Hopkins University School of Medicine, Baltimore, MD, USA"],"raw_orcid":"https://orcid.org/0009-0004-9247-4349","affiliations":[{"raw_affiliation_string":"Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","institution_ids":["https://openalex.org/I145311948"]},{"raw_affiliation_string":"Department of Molecular Biology and Genetics, https://ror.org/00za53h95Johns Hopkins University School of Medicine, Baltimore, MD, USA","institution_ids":["https://openalex.org/I145311948","https://openalex.org/I2799853436"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5066990503","display_name":"Taylor Anglen","orcid":"https://orcid.org/0000-0003-4451-3046","observed_orcids":["https://orcid.org/0000-0003-4451-3046"]},"institutions":[{"id":"https://openalex.org/I170897317","display_name":"Duke University","ror":"https://ror.org/00py81415","country_code":"US","type":"education","lineage":["https://openalex.org/I170897317"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Taylor Anglen","raw_affiliation_strings":["Duke University 2 Department of Biomedical Engineering, , Durham, NC,","Department of Biomedical Engineering, https://ror.org/00py81415Duke University, Durham, NC, USA"],"raw_orcid":"https://orcid.org/0000-0003-4451-3046","affiliations":[{"raw_affiliation_string":"Duke University 2 Department of Biomedical Engineering, , Durham, NC,","institution_ids":["https://openalex.org/I170897317"]},{"raw_affiliation_string":"Department of Biomedical Engineering, https://ror.org/00py81415Duke University, Durham, NC, USA","institution_ids":["https://openalex.org/I170897317"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5025431352","display_name":"Margaret A. Strong","orcid":"https://orcid.org/0000-0002-0055-6573","observed_orcids":["https://orcid.org/0000-0002-0055-6573"]},"institutions":[{"id":"https://openalex.org/I145311948","display_name":"Johns Hopkins University","ror":"https://ror.org/00za53h95","country_code":"US","type":"education","lineage":["https://openalex.org/I145311948"]},{"id":"https://openalex.org/I2799853436","display_name":"Johns Hopkins Medicine","ror":"https://ror.org/037zgn354","country_code":"US","type":"healthcare","lineage":["https://openalex.org/I2799853436"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Margaret A. Strong","raw_affiliation_strings":["Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","Department of Molecular Biology and Genetics, https://ror.org/00za53h95Johns Hopkins University School of Medicine, Baltimore, MD, USA"],"raw_orcid":"https://orcid.org/0000-0002-0055-6573","affiliations":[{"raw_affiliation_string":"Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","institution_ids":["https://openalex.org/I145311948"]},{"raw_affiliation_string":"Department of Molecular Biology and Genetics, https://ror.org/00za53h95Johns Hopkins University School of Medicine, Baltimore, MD, USA","institution_ids":["https://openalex.org/I145311948","https://openalex.org/I2799853436"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5062562116","display_name":"Colin R. Gliech","orcid":"https://orcid.org/0000-0002-5410-1718","observed_orcids":["https://orcid.org/0000-0002-5410-1718"]},"institutions":[{"id":"https://openalex.org/I145311948","display_name":"Johns Hopkins University","ror":"https://ror.org/00za53h95","country_code":"US","type":"education","lineage":["https://openalex.org/I145311948"]},{"id":"https://openalex.org/I2799853436","display_name":"Johns Hopkins Medicine","ror":"https://ror.org/037zgn354","country_code":"US","type":"healthcare","lineage":["https://openalex.org/I2799853436"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Colin R. Gliech","raw_affiliation_strings":["Johns Hopkins University School of Medicine 3 Department of Rheumatology, , Baltimore, MD,","Department of Rheumatology, https://ror.org/00za53h95Johns Hopkins University School of Medicine, Baltimore, MD, USA"],"raw_orcid":"https://orcid.org/0000-0002-5410-1718","affiliations":[{"raw_affiliation_string":"Johns Hopkins University School of Medicine 3 Department of Rheumatology, , Baltimore, MD,","institution_ids":["https://openalex.org/I145311948","https://openalex.org/I2799853436"]},{"raw_affiliation_string":"Department of Rheumatology, https://ror.org/00za53h95Johns Hopkins University School of Medicine, Baltimore, MD, USA","institution_ids":["https://openalex.org/I145311948","https://openalex.org/I2799853436"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5051074925","display_name":"Cayla E Jewett","orcid":"https://orcid.org/0000-0002-8406-0814","observed_orcids":["https://orcid.org/0000-0002-8406-0814"]},"institutions":[{"id":"https://openalex.org/I145311948","display_name":"Johns Hopkins University","ror":"https://ror.org/00za53h95","country_code":"US","type":"education","lineage":["https://openalex.org/I145311948"]},{"id":"https://openalex.org/I2799853436","display_name":"Johns Hopkins Medicine","ror":"https://ror.org/037zgn354","country_code":"US","type":"healthcare","lineage":["https://openalex.org/I2799853436"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Cayla E. Jewett","raw_affiliation_strings":["Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","Department of Molecular Biology and Genetics, https://ror.org/00za53h95Johns Hopkins University School of Medicine, Baltimore, MD, USA"],"raw_orcid":"https://orcid.org/0000-0002-8406-0814","affiliations":[{"raw_affiliation_string":"Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","institution_ids":["https://openalex.org/I145311948"]},{"raw_affiliation_string":"Department of Molecular Biology and Genetics, https://ror.org/00za53h95Johns Hopkins University School of Medicine, Baltimore, MD, USA","institution_ids":["https://openalex.org/I145311948","https://openalex.org/I2799853436"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5090302884","display_name":"Anoek Friskes","orcid":"https://orcid.org/0000-0002-9845-3715","observed_orcids":["https://orcid.org/0000-0002-9845-3715"]},"institutions":[{"id":"https://openalex.org/I2898336195","display_name":"The Netherlands Cancer Institute","ror":"https://ror.org/03xqtf034","country_code":"NL","type":"healthcare","lineage":["https://openalex.org/I2898336195"]}],"countries":["NL"],"is_corresponding":false,"raw_author_name":"Anoek Friskes","raw_affiliation_strings":["Oncode Institute, The Netherlands Cancer Institute 4 Division of Cell Biology, , Amsterdam,","Division of Cell Biology, https://ror.org/01n92vv28Oncode Institute, The Netherlands Cancer Institute, Amsterdam, Netherlands"],"raw_orcid":"https://orcid.org/0000-0002-9845-3715","affiliations":[{"raw_affiliation_string":"Oncode Institute, The Netherlands Cancer Institute 4 Division of Cell Biology, , Amsterdam,","institution_ids":["https://openalex.org/I2898336195"]},{"raw_affiliation_string":"Division of Cell Biology, https://ror.org/01n92vv28Oncode Institute, The Netherlands Cancer Institute, Amsterdam, Netherlands","institution_ids":["https://openalex.org/I2898336195"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5046322500","display_name":"Thao P. Phan","orcid":"https://orcid.org/0000-0003-0389-7127","observed_orcids":["https://orcid.org/0000-0003-0389-7127"]},"institutions":[{"id":"https://openalex.org/I180670191","display_name":"University of California, San Francisco","ror":"https://ror.org/043mz5j54","country_code":"US","type":"education","lineage":["https://openalex.org/I180670191"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Thao P. Phan","raw_affiliation_strings":["Cardiovascular Research Institute, University of California, San Francisco 5 Department of Biochemistry and Biophysics, , San Francisco, CA,","Department of Biochemistry and Biophysics, https://ror.org/043mz5j54Cardiovascular Research Institute, University of California, San Francisco, San Francisco, CA, USA"],"raw_orcid":"https://orcid.org/0000-0003-0389-7127","affiliations":[{"raw_affiliation_string":"Cardiovascular Research Institute, University of California, San Francisco 5 Department of Biochemistry and Biophysics, , San Francisco, CA,","institution_ids":["https://openalex.org/I180670191"]},{"raw_affiliation_string":"Department of Biochemistry and Biophysics, https://ror.org/043mz5j54Cardiovascular Research Institute, University of California, San Francisco, San Francisco, CA, USA","institution_ids":["https://openalex.org/I180670191"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5116423016","display_name":"Zachary Nicholas","orcid":"https://orcid.org/0000-0002-7495-4080","observed_orcids":["https://orcid.org/0000-0002-7495-4080"]},"institutions":[{"id":"https://openalex.org/I145311948","display_name":"Johns Hopkins University","ror":"https://ror.org/00za53h95","country_code":"US","type":"education","lineage":["https://openalex.org/I145311948"]},{"id":"https://openalex.org/I2799853436","display_name":"Johns Hopkins Medicine","ror":"https://ror.org/037zgn354","country_code":"US","type":"healthcare","lineage":["https://openalex.org/I2799853436"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Zachary Nicholas","raw_affiliation_strings":["Johns Hopkins University School of Medicine 6 Department of Neuroscience, , Baltimore, MD,","Department of Neuroscience, https://ror.org/00za53h95Johns Hopkins University School of Medicine, Baltimore, MD, USA"],"raw_orcid":"https://orcid.org/0000-0002-7495-4080","affiliations":[{"raw_affiliation_string":"Johns Hopkins University School of Medicine 6 Department of Neuroscience, , Baltimore, MD,","institution_ids":["https://openalex.org/I145311948","https://openalex.org/I2799853436"]},{"raw_affiliation_string":"Department of Neuroscience, https://ror.org/00za53h95Johns Hopkins University School of Medicine, Baltimore, MD, USA","institution_ids":["https://openalex.org/I145311948","https://openalex.org/I2799853436"]}]},{"author_position":"last","author":{"id":"https://openalex.org/A5001482747","display_name":"Andrew Jon Holland","orcid":"https://orcid.org/0000-0003-3728-6367","observed_orcids":["https://orcid.org/0000-0003-3728-6367"]},"institutions":[{"id":"https://openalex.org/I145311948","display_name":"Johns Hopkins University","ror":"https://ror.org/00za53h95","country_code":"US","type":"education","lineage":["https://openalex.org/I145311948"]},{"id":"https://openalex.org/I2799853436","display_name":"Johns Hopkins Medicine","ror":"https://ror.org/037zgn354","country_code":"US","type":"healthcare","lineage":["https://openalex.org/I2799853436"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Andrew J. Holland","raw_affiliation_strings":["Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","Department of Molecular Biology and Genetics, https://ror.org/00za53h95Johns Hopkins University School of Medicine, Baltimore, MD, USA"],"raw_orcid":"https://orcid.org/0000-0003-3728-6367","affiliations":[{"raw_affiliation_string":"Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","institution_ids":["https://openalex.org/I145311948"]},{"raw_affiliation_string":"Department of Molecular Biology and Genetics, https://ror.org/00za53h95Johns Hopkins University School of Medicine, Baltimore, MD, USA","institution_ids":["https://openalex.org/I145311948","https://openalex.org/I2799853436"]}]}],"institutions":[],"countries_distinct_count":2,"institutions_distinct_count":5,"corresponding_author_ids":["https://openalex.org/A5037731819"],"corresponding_institution_ids":["https://openalex.org/I145311948","https://openalex.org/I2799853436"],"apc_list":null,"apc_paid":null,"fwci":2.9928,"has_fulltext":false,"cited_by_count":9,"citation_normalized_percentile":{"value":0.91441079,"is_in_top_1_percent":false,"is_in_top_10_percent":true},"cited_by_percentile_year":{"min":93,"max":98},"biblio":{"volume":"224","issue":"4","first_page":null,"last_page":null},"is_retracted":false,"is_paratext":false,"is_xpac":false,"primary_topic":{"id":"https://openalex.org/T11412","display_name":"Genetic and Kidney Cyst Diseases","score":0.9955999851226807,"subfield":{"id":"https://openalex.org/subfields/1311","display_name":"Genetics"},"field":{"id":"https://openalex.org/fields/13","display_name":"Biochemistry, Genetics and Molecular Biology"},"domain":{"id":"https://openalex.org/domains/1","display_name":"Life Sciences"}},"topics":[{"id":"https://openalex.org/T11412","display_name":"Genetic and Kidney Cyst Diseases","score":0.9955999851226807,"subfield":{"id":"https://openalex.org/subfields/1311","display_name":"Genetics"},"field":{"id":"https://openalex.org/fields/13","display_name":"Biochemistry, Genetics and Molecular Biology"},"domain":{"id":"https://openalex.org/domains/1","display_name":"Life Sciences"}},{"id":"https://openalex.org/T10665","display_name":"Cystic Fibrosis Research Advances","score":0.002300000051036477,"subfield":{"id":"https://openalex.org/subfields/2740","display_name":"Pulmonary and Respiratory Medicine"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},{"id":"https://openalex.org/T10492","display_name":"Microtubule and mitosis dynamics","score":0.0003000000142492354,"subfield":{"id":"https://openalex.org/subfields/1307","display_name":"Cell Biology"},"field":{"id":"https://openalex.org/fields/13","display_name":"Biochemistry, Genetics and Molecular Biology"},"domain":{"id":"https://openalex.org/domains/1","display_name":"Life Sciences"}}],"keywords":[{"id":"https://openalex.org/keywords/hydrolethalus-syndrome","display_name":"hydrolethalus syndrome","score":0.9940000176429749},{"id":"https://openalex.org/keywords/ciliopathy","display_name":"ciliopathies","score":0.8650000095367432},{"id":"https://openalex.org/keywords/ciliogenesis","display_name":"ciliogenesis","score":0.8989999890327454},{"id":"https://openalex.org/keywords/mouse-model","display_name":"mouse model","score":0.6610000133514404}],"concepts":[{"id":"https://openalex.org/C195267511","wikidata":"https://www.wikidata.org/wiki/Q5119969","display_name":"Ciliogenesis","level":3,"score":0.9411022067070007},{"id":"https://openalex.org/C48342290","wikidata":"https://www.wikidata.org/wiki/Q178355","display_name":"Centriole","level":3,"score":0.9014785885810852},{"id":"https://openalex.org/C100094513","wikidata":"https://www.wikidata.org/wiki/Q180436","display_name":"Cilium","level":2,"score":0.8852156400680542},{"id":"https://openalex.org/C86803240","wikidata":"https://www.wikidata.org/wiki/Q420","display_name":"Biology","level":0,"score":0.6720442175865173},{"id":"https://openalex.org/C95444343","wikidata":"https://www.wikidata.org/wiki/Q7141","display_name":"Cell biology","level":1,"score":0.6552455425262451},{"id":"https://openalex.org/C127716648","wikidata":"https://www.wikidata.org/wiki/Q104053","display_name":"Phenotype","level":3,"score":0.576048731803894},{"id":"https://openalex.org/C501734568","wikidata":"https://www.wikidata.org/wiki/Q42918","display_name":"Mutation","level":3,"score":0.5262357592582703},{"id":"https://openalex.org/C2780561973","wikidata":"https://www.wikidata.org/wiki/Q203031","display_name":"Ciliopathy","level":4,"score":0.49375423789024353},{"id":"https://openalex.org/C10447061","wikidata":"https://www.wikidata.org/wiki/Q190769","display_name":"Centrosome","level":4,"score":0.44758760929107666},{"id":"https://openalex.org/C54355233","wikidata":"https://www.wikidata.org/wiki/Q7162","display_name":"Genetics","level":1,"score":0.4155426025390625},{"id":"https://openalex.org/C20418707","wikidata":"https://www.wikidata.org/wiki/Q189933","display_name":"Microtubule","level":2,"score":0.3472692370414734},{"id":"https://openalex.org/C104317684","wikidata":"https://www.wikidata.org/wiki/Q7187","display_name":"Gene","level":2,"score":0.08472472429275513},{"id":"https://openalex.org/C29537977","wikidata":"https://www.wikidata.org/wiki/Q188941","display_name":"Cell cycle","level":3,"score":0.07390832901000977}],"mesh":[],"locations_count":7,"locations":[{"id":"doi:10.1083/jcb.202403022","doi":"https://doi.org/10.1083/jcb.202403022","is_oa":false,"landing_page_url":"https://doi.org/10.1083/jcb.202403022","pdf_url":null,"source":{"id":"https://openalex.org/S96410829","display_name":"The Journal of Cell Biology","issn_l":"0021-9525","issn":["0021-9525","1540-8140"],"is_oa":false,"is_in_doaj":false,"is_core":true,"listed_in":["anvur-scientific","cwts-core","dongbi-a","doyens","jufo-3","ki-jl-2","medline","norway-2","poland-140","russia-white-list-1"],"host_organization":"https://openalex.org/P4310315676","host_organization_name":"Rockefeller University Press","host_organization_lineage":["https://openalex.org/P4310315676"],"host_organization_lineage_names":["Rockefeller University Press"],"type":"journal"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Journal of Cell Biology","raw_type":"journal-article"},{"id":"doi:10.1101/2024.03.06.583733","doi":"https://doi.org/10.1101/2024.03.06.583733","is_oa":true,"landing_page_url":"https://doi.org/10.1101/2024.03.06.583733","pdf_url":"https://www.biorxiv.org/content/biorxiv/early/2024/03/06/2024.03.06.583733.full.pdf","source":{"id":"https://openalex.org/S4306402567","display_name":"bioRxiv (Cold Spring Harbor Laboratory)","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I2750212522","host_organization_name":"Cold Spring Harbor Laboratory","host_organization_lineage":["https://openalex.org/I2750212522"],"host_organization_lineage_names":[],"type":"repository"},"license":"cc-by-nc-nd","license_id":"https://openalex.org/licenses/cc-by-nc-nd","version":"acceptedVersion","is_accepted":true,"is_published":false,"raw_source_name":"bioRxiv","raw_type":"posted-content"},{"id":"pmh:oai:europepmc.org:11722863","doi":null,"is_oa":true,"landing_page_url":"https://www.ncbi.nlm.nih.gov/pmc/articles/11864076","pdf_url":null,"source":{"id":"https://openalex.org/S4306400806","display_name":"Europe PMC (PubMed Central)","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I1303153112","host_organization_name":"European Bioinformatics Institute","host_organization_lineage":["https://openalex.org/I1303153112"],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":null,"raw_type":"Text"},{"id":"pmid:38496445","doi":null,"is_oa":false,"landing_page_url":"https://pubmed.ncbi.nlm.nih.gov/38496445","pdf_url":null,"source":{"id":"https://openalex.org/S4306525036","display_name":"PubMed","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I1299303238","host_organization_name":"National Institutes of Health","host_organization_lineage":["https://openalex.org/I1299303238"],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"bioRxiv : the preprint server for biology","raw_type":"Preprint"},{"id":"pmid:40009365","doi":null,"is_oa":false,"landing_page_url":"https://pubmed.ncbi.nlm.nih.gov/40009365","pdf_url":null,"source":{"id":"https://openalex.org/S4306525036","display_name":"PubMed","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I1299303238","host_organization_name":"National Institutes of Health","host_organization_lineage":["https://openalex.org/I1299303238"],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"The Journal of cell biology","raw_type":"Journal Article"},{"id":"pmh:oai:pubmedcentral.nih.gov:11864076","doi":null,"is_oa":true,"landing_page_url":"https://pmc.ncbi.nlm.nih.gov/articles/PMC11864076/","pdf_url":null,"source":{"id":"https://openalex.org/S2764455111","display_name":"PubMed Central","issn_l":null,"issn":null,"is_oa":true,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I1299303238","host_organization_name":"National Institutes of Health","host_organization_lineage":["https://openalex.org/I1299303238"],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"J Cell Biol","raw_type":"Text"},{"id":"pmh:oai:pubmedcentral.nih.gov:10942441","doi":null,"is_oa":true,"landing_page_url":"https://www.ncbi.nlm.nih.gov/pmc/articles/10942441","pdf_url":null,"source":{"id":"https://openalex.org/S2764455111","display_name":"PubMed Central","issn_l":null,"issn":null,"is_oa":true,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I1299303238","host_organization_name":"National Institutes of Health","host_organization_lineage":["https://openalex.org/I1299303238"],"host_organization_lineage_names":[],"type":"repository"},"license":"cc-by-nc-nd","license_id":"https://openalex.org/licenses/cc-by-nc-nd","version":"submittedVersion","is_accepted":false,"is_published":false,"raw_source_name":"bioRxiv","raw_type":"Text"}],"best_oa_location":{"id":"doi:10.1101/2024.03.06.583733","doi":"https://doi.org/10.1101/2024.03.06.583733","is_oa":true,"landing_page_url":"https://doi.org/10.1101/2024.03.06.583733","pdf_url":"https://www.biorxiv.org/content/biorxiv/early/2024/03/06/2024.03.06.583733.full.pdf","source":{"id":"https://openalex.org/S4306402567","display_name":"bioRxiv (Cold Spring Harbor Laboratory)","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I2750212522","host_organization_name":"Cold Spring Harbor Laboratory","host_organization_lineage":["https://openalex.org/I2750212522"],"host_organization_lineage_names":[],"type":"repository"},"license":"cc-by-nc-nd","license_id":"https://openalex.org/licenses/cc-by-nc-nd","version":"acceptedVersion","is_accepted":true,"is_published":false,"raw_source_name":"bioRxiv","raw_type":"posted-content"},"sustainable_development_goals":[{"id":"https://metadata.un.org/sdg/3","score":0.46810001134872437,"display_name":"Good health and well-being"}],"study_designs":[],"awards":[{"id":"https://openalex.org/G3237617285","display_name":null,"funder_award_id":"DRG-2478-22","funder_id":"https://openalex.org/F4320306271","funder_display_name":"Damon Runyon Cancer Research Foundation"},{"id":"https://openalex.org/G5440157153","display_name":"Molecular mechanism that suppresses the proliferation of cells with supernumerary centrioles","funder_award_id":"R01GM133897","funder_id":"https://openalex.org/F4320337354","funder_display_name":"National Institute of General Medical Sciences"},{"id":"https://openalex.org/G6411239323","display_name":"Centrosome biogenesis and copy number control","funder_award_id":"R01GM114119","funder_id":"https://openalex.org/F4320337354","funder_display_name":"National Institute of General Medical Sciences"},{"id":"https://openalex.org/G9000887295","display_name":"The role of TRIM37 in driving tumorigenesis and cancer-specific vulnerability to PLK4 inhibition","funder_award_id":"R01CA266199","funder_id":"https://openalex.org/F4320337351","funder_display_name":"National Cancer Institute"}],"funders":[{"id":"https://openalex.org/F4320306271","display_name":"Damon Runyon Cancer Research Foundation","ror":"https://ror.org/01gd7b947"},{"id":"https://openalex.org/F4320332161","display_name":"National Institutes of Health","ror":"https://ror.org/01cwqze88"},{"id":"https://openalex.org/F4320337351","display_name":"National Cancer Institute","ror":"https://ror.org/040gcmg81"},{"id":"https://openalex.org/F4320337354","display_name":"National Institute of General Medical Sciences","ror":"https://ror.org/04q48ey07"}],"has_content":{"pdf":true,"grobid_xml":true},"content_urls":{"pdf":"https://content.openalex.org/works/W4407946372.pdf","grobid_xml":"https://content.openalex.org/works/W4407946372.grobid-xml"},"referenced_works_count":66,"referenced_works":["https://openalex.org/W1976311440","https://openalex.org/W1977207226","https://openalex.org/W1985622227","https://openalex.org/W1995891092","https://openalex.org/W2004140454","https://openalex.org/W2016052475","https://openalex.org/W2024687001","https://openalex.org/W2028544866","https://openalex.org/W2042378344","https://openalex.org/W2051707571","https://openalex.org/W2055491482","https://openalex.org/W2080823011","https://openalex.org/W2090966394","https://openalex.org/W2098147370","https://openalex.org/W2099428434","https://openalex.org/W2103390035","https://openalex.org/W2106208777","https://openalex.org/W2109700293","https://openalex.org/W2117401183","https://openalex.org/W2128126810","https://openalex.org/W2130117786","https://openalex.org/W2144833749","https://openalex.org/W2146703382","https://openalex.org/W2159243681","https://openalex.org/W2165775690","https://openalex.org/W2167279371","https://openalex.org/W2169614209","https://openalex.org/W2179957908","https://openalex.org/W2233747175","https://openalex.org/W2347284578","https://openalex.org/W2395055872","https://openalex.org/W2398230902","https://openalex.org/W2404094468","https://openalex.org/W2422201302","https://openalex.org/W2515973749","https://openalex.org/W2582256818","https://openalex.org/W2606833132","https://openalex.org/W2734540391","https://openalex.org/W2785424791","https://openalex.org/W2789611794","https://openalex.org/W2804603137","https://openalex.org/W2890354249","https://openalex.org/W2902929870","https://openalex.org/W2908004295","https://openalex.org/W2938261111","https://openalex.org/W2945568996","https://openalex.org/W2949627826","https://openalex.org/W3016237797","https://openalex.org/W3038485847","https://openalex.org/W3087159146","https://openalex.org/W3100603793","https://openalex.org/W3106842458","https://openalex.org/W3107088376","https://openalex.org/W3117988574","https://openalex.org/W3136704442","https://openalex.org/W3175217268","https://openalex.org/W3207344627","https://openalex.org/W4210754131","https://openalex.org/W4223442630","https://openalex.org/W4283725346","https://openalex.org/W4291899373","https://openalex.org/W4293828036","https://openalex.org/W4296788965","https://openalex.org/W4388453840","https://openalex.org/W4391256130","https://openalex.org/W4393096745"],"related_works":["https://openalex.org/W2103390035","https://openalex.org/W3016237797","https://openalex.org/W2606833132","https://openalex.org/W7223020321","https://openalex.org/W2109700293","https://openalex.org/W2515973749","https://openalex.org/W1967146176","https://openalex.org/W2130117786","https://openalex.org/W3175217268","https://openalex.org/W3173666353"],"abstract_inverted_index":{"Hydrolethalus":[0],"Syndrome":[1],"(HLS)":[2],"is":[3,28,79],"a":[4,70],"lethal,":[5],"autosomal":[6],"recessive":[7],"ciliopathy":[8],"caused":[9,68,129],"by":[10,69,84,130],"the":[11,14,23,37,54,82,96,104,117,131],"mutation":[12,40,102,133],"of":[13,26,53,72,106,116],"conserved":[15],"centriole":[16,73,83,90,118,127],"protein":[17],"HYLS1.":[18],"However,":[19],"how":[20],"HYLS1":[21,38,78,107,112,132],"facilitates":[22],"centriole-based":[24],"templating":[25],"cilia":[27,64],"poorly":[29],"understood.":[30],"Here,":[31],"we":[32],"show":[33,76],"that":[34,49,77,94,123],"mice":[35],"harboring":[36],"disease":[39],"die":[41],"shortly":[42],"after":[43],"birth":[44],"and":[45,66,86,114,136],"exhibit":[46],"developmental":[47],"defects":[48,62,125],"recapitulate":[50],"several":[51],"manifestations":[52],"human":[55],"disease.":[56],"These":[57],"phenotypes":[58],"arise":[59],"from":[60],"tissue-specific":[61,124],"in":[63,126],"assembly":[65],"function":[67],"loss":[71],"integrity.":[74],"We":[75,121],"recruited":[80],"to":[81,88,111],"CEP120":[85,109],"functions":[87],"recruit":[89],"inner":[91],"scaffold":[92],"proteins":[93],"stabilize":[95],"centriolar":[97],"microtubule":[98],"wall.":[99],"The":[100],"HLS":[101,138],"disrupts":[103],"interaction":[105],"with":[108],"leading":[110],"displacement":[113],"degeneration":[115],"distal":[119],"end.":[120],"propose":[122],"integrity":[128],"prevent":[134],"ciliogenesis":[135],"drive":[137],"phenotypes.":[139]},"counts_by_year":[{"year":2026,"cited_by_count":4},{"year":2025,"cited_by_count":3},{"year":2024,"cited_by_count":2}],"updated_date":"2026-11-08T12:12:28.789561","created_date":"2025-02-26T00:00:00"}