{"id":"https://openalex.org/W4397046408","doi":"https://doi.org/10.1681/asn.20223311s1421c","title":"Human Collecting Duct Tubuloids of Principal Cells Derived From Ureteric Bud Organoids as Differentiated Cell Models for Physiology and Disease Modelling","display_name":"Human Collecting Duct Tubuloids of Principal Cells Derived From Ureteric Bud Organoids as Differentiated Cell Models for Physiology and Disease Modelling","publication_year":2022,"publication_date":"2022-11-01","ids":{"openalex":"https://openalex.org/W4397046408","doi":"https://doi.org/10.1681/asn.20223311s1421c"},"language":"en","primary_location":{"id":"doi:10.1681/asn.20223311s1421c","doi":"https://doi.org/10.1681/asn.20223311s1421c","is_oa":false,"landing_page_url":"https://doi.org/10.1681/asn.20223311s1421c","pdf_url":null,"source":{"id":"https://openalex.org/S115693375","display_name":"Journal of the American Society of Nephrology","issn_l":"1046-6673","issn":["1046-6673","1533-3450"],"is_oa":false,"is_in_doaj":false,"is_core":true,"listed_in":["anvur-scientific","cwts-core","dongbi-a","doyens","jufo-3","ki-jl-3","medline","norway-2","poland-200","russia-white-list-1","vabb-shw"],"host_organization":"https://openalex.org/P4310315620","host_organization_name":"American Society of Nephrology","host_organization_lineage":["https://openalex.org/P4310315620"],"host_organization_lineage_names":["American Society of Nephrology"],"type":"journal"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Journal of the American Society of Nephrology","raw_type":"journal-article"},"volume":null,"type":"conference-abstract","is_traditional":false,"indexed_in":["crossref"],"open_access":{"is_oa":false,"oa_status":"closed","oa_url":null,"any_repository_has_fulltext":false},"authorships":[{"author_position":"first","author":{"id":"https://openalex.org/A5054938229","display_name":"Annarita Di Mise","orcid":"https://orcid.org/0000-0002-8132-4917","observed_orcids":["https://orcid.org/0000-0002-8132-4917"]},"institutions":[{"id":"https://openalex.org/I5561750","display_name":"University of Bari Aldo Moro","ror":"https://ror.org/027ynra39","country_code":"IT","type":"education","lineage":["https://openalex.org/I5561750"]}],"countries":["IT"],"is_corresponding":false,"raw_author_name":"Annarita Di Mise","raw_affiliation_strings":["Universita degli Studi di Bari Aldo Moro, Bari, Italy"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Universita degli Studi di Bari Aldo Moro, Bari, Italy","institution_ids":["https://openalex.org/I5561750"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5022146957","display_name":"Kyle W. McCracken","orcid":"https://orcid.org/0000-0001-7650-5513","observed_orcids":["https://orcid.org/0000-0001-7650-5513"]},"institutions":[{"id":"https://openalex.org/I1288882113","display_name":"Boston Children's Hospital","ror":"https://ror.org/00dvg7y05","country_code":"US","type":"healthcare","lineage":["https://openalex.org/I1288882113"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Kyle McCracken","raw_affiliation_strings":["Boston Children's Hospital, Boston, MA"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Boston Children's Hospital, Boston, MA","institution_ids":["https://openalex.org/I1288882113"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5014987135","display_name":"Ankit Bharat Patel","orcid":"https://orcid.org/0000-0001-6812-1464","observed_orcids":["https://orcid.org/0000-0001-6812-1464"]},"institutions":[{"id":"https://openalex.org/I1283280774","display_name":"Brigham and Women's Hospital","ror":"https://ror.org/04b6nzv94","country_code":"US","type":"healthcare","lineage":["https://openalex.org/I121934306","https://openalex.org/I1283280774","https://openalex.org/I48633490"]},{"id":"https://openalex.org/I61970326","display_name":"Vertex Pharmaceuticals (United States)","ror":"https://ror.org/00anb1726","country_code":"US","type":"company","lineage":["https://openalex.org/I61970326"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Ankit B. Patel","raw_affiliation_strings":["Brigham and Women's Hospital, Boston, MA","Vertex Pharmaceuticals Incorporated, Boston, MA"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Brigham and Women's Hospital, Boston, MA","institution_ids":["https://openalex.org/I1283280774"]},{"raw_affiliation_string":"Vertex Pharmaceuticals Incorporated, Boston, MA","institution_ids":["https://openalex.org/I61970326"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5016382712","display_name":"Giovanna Valenti","orcid":"https://orcid.org/0000-0003-0233-0778","observed_orcids":["https://orcid.org/0000-0003-0233-0778"]},"institutions":[{"id":"https://openalex.org/I5561750","display_name":"University of Bari Aldo Moro","ror":"https://ror.org/027ynra39","country_code":"IT","type":"education","lineage":["https://openalex.org/I5561750"]}],"countries":["IT"],"is_corresponding":false,"raw_author_name":"Giovanna Valenti","raw_affiliation_strings":["Universita degli Studi di Bari Aldo Moro, Bari, Italy"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Universita degli Studi di Bari Aldo Moro, Bari, Italy","institution_ids":["https://openalex.org/I5561750"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5080273574","display_name":"Takaharu Ichimura","orcid":"https://orcid.org/0000-0002-3106-5224","observed_orcids":["https://orcid.org/0000-0002-3106-5224"]},"institutions":[{"id":"https://openalex.org/I1283280774","display_name":"Brigham and Women's Hospital","ror":"https://ror.org/04b6nzv94","country_code":"US","type":"healthcare","lineage":["https://openalex.org/I121934306","https://openalex.org/I1283280774","https://openalex.org/I48633490"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Takaharu Ichimura","raw_affiliation_strings":["Brigham and Women's Hospital, Boston, MA"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Brigham and Women's Hospital, Boston, MA","institution_ids":["https://openalex.org/I1283280774"]}]},{"author_position":"last","author":{"id":"https://openalex.org/A5089781525","display_name":"Joseph Vincent Bonventre","orcid":"https://orcid.org/0000-0001-7144-386X","observed_orcids":["https://orcid.org/0000-0001-7144-386X"]},"institutions":[{"id":"https://openalex.org/I1283280774","display_name":"Brigham and Women's Hospital","ror":"https://ror.org/04b6nzv94","country_code":"US","type":"healthcare","lineage":["https://openalex.org/I121934306","https://openalex.org/I1283280774","https://openalex.org/I48633490"]},{"id":"https://openalex.org/I136199984","display_name":"Harvard University","ror":"https://ror.org/03vek6s52","country_code":"US","type":"education","lineage":["https://openalex.org/I136199984"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Joseph V. Bonventre","raw_affiliation_strings":["Division of Renal Medicine, Department of Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Division of Renal Medicine, Department of Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA","institution_ids":["https://openalex.org/I1283280774","https://openalex.org/I136199984"]}]}],"institutions":[],"countries_distinct_count":2,"institutions_distinct_count":5,"corresponding_author_ids":[],"corresponding_institution_ids":[],"apc_list":null,"apc_paid":null,"fwci":null,"has_fulltext":false,"cited_by_count":0,"citation_normalized_percentile":null,"cited_by_percentile_year":null,"biblio":{"volume":"33","issue":"11S","first_page":"421","last_page":"421"},"is_retracted":false,"is_paratext":false,"is_xpac":false,"primary_topic":{"id":"https://openalex.org/T10724","display_name":"Ion Transport and Channel Regulation","score":0.9711999893188477,"subfield":{"id":"https://openalex.org/subfields/1312","display_name":"Molecular 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"}},"topics":[{"id":"https://openalex.org/T10724","display_name":"Ion Transport and Channel Regulation","score":0.9711999893188477,"subfield":{"id":"https://openalex.org/subfields/1312","display_name":"Molecular 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"}},{"id":"https://openalex.org/T12140","display_name":"Renal and related cancers","score":0.008200000040233135,"subfield":{"id":"https://openalex.org/subfields/2730","display_name":"Oncology"},"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/T10505","display_name":"Pluripotent Stem Cells Research","score":0.004900000058114529,"subfield":{"id":"https://openalex.org/subfields/1312","display_name":"Molecular 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/collecting-duct-principal-cells","display_name":"collecting duct principal cells","score":0.7549999952316284},{"id":"https://openalex.org/keywords/aquaporin-2","display_name":"aquaporin-2","score":0.6650000214576721},{"id":"https://openalex.org/keywords/vasopressin-v2-receptor","display_name":"V2 receptor","score":0.656000018119812},{"id":"https://openalex.org/keywords/desmopressin","display_name":"desmopressin","score":0.902999997138977},{"id":"https://openalex.org/keywords/avpr2","display_name":"AVPR2","score":0.6869999766349792},{"id":"https://openalex.org/keywords/pc2","display_name":"PC2","score":0.9110000133514404},{"id":"https://openalex.org/keywords/autosomal-dominant-polycystic-kidney-disease","display_name":"autosomal dominant polycystic kidney disease","score":0.9599999785423279},{"id":"https://openalex.org/keywords/nephrogenic-diabetes-insipidus","display_name":"nephrogenic diabetes insipidus","score":0.9670000076293945},{"id":"https://openalex.org/keywords/transepithelial-potential","display_name":"transepithelial potential","score":0.7059999704360962},{"id":"https://openalex.org/keywords/water-permeability","display_name":"water permeability","score":0.4490000009536743}],"concepts":[{"id":"https://openalex.org/C31695470","wikidata":"https://www.wikidata.org/wiki/Q11293125","display_name":"Organoid","level":2,"score":0.8189985752105713},{"id":"https://openalex.org/C86803240","wikidata":"https://www.wikidata.org/wiki/Q420","display_name":"Biology","level":0,"score":0.47896721959114075},{"id":"https://openalex.org/C1491633281","wikidata":"https://www.wikidata.org/wiki/Q7868","display_name":"Cell","level":2,"score":0.43615198135375977},{"id":"https://openalex.org/C2779134260","wikidata":"https://www.wikidata.org/wiki/Q12136","display_name":"Disease","level":2,"score":0.42660245299339294},{"id":"https://openalex.org/C95444343","wikidata":"https://www.wikidata.org/wiki/Q7141","display_name":"Cell biology","level":1,"score":0.3591424226760864},{"id":"https://openalex.org/C142724271","wikidata":"https://www.wikidata.org/wiki/Q7208","display_name":"Pathology","level":1,"score":0.3311719298362732},{"id":"https://openalex.org/C71924100","wikidata":"https://www.wikidata.org/wiki/Q11190","display_name":"Medicine","level":0,"score":0.2633005380630493},{"id":"https://openalex.org/C55493867","wikidata":"https://www.wikidata.org/wiki/Q7094","display_name":"Biochemistry","level":1,"score":0.05715322494506836}],"mesh":[],"locations_count":1,"locations":[{"id":"doi:10.1681/asn.20223311s1421c","doi":"https://doi.org/10.1681/asn.20223311s1421c","is_oa":false,"landing_page_url":"https://doi.org/10.1681/asn.20223311s1421c","pdf_url":null,"source":{"id":"https://openalex.org/S115693375","display_name":"Journal of the American Society of Nephrology","issn_l":"1046-6673","issn":["1046-6673","1533-3450"],"is_oa":false,"is_in_doaj":false,"is_core":true,"listed_in":["anvur-scientific","cwts-core","dongbi-a","doyens","jufo-3","ki-jl-3","medline","norway-2","poland-200","russia-white-list-1","vabb-shw"],"host_organization":"https://openalex.org/P4310315620","host_organization_name":"American Society of Nephrology","host_organization_lineage":["https://openalex.org/P4310315620"],"host_organization_lineage_names":["American Society of Nephrology"],"type":"journal"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Journal of the American Society of Nephrology","raw_type":"journal-article"}],"best_oa_location":null,"sustainable_development_goals":[{"score":0.9391000270843506,"display_name":"Good health and well-being","id":"https://metadata.un.org/sdg/3"}],"study_designs":[],"awards":[],"funders":[],"has_content":{"pdf":false,"grobid_xml":false},"content_urls":null,"referenced_works_count":0,"referenced_works":[],"related_works":["https://openalex.org/W4307658815","https://openalex.org/W4403832858","https://openalex.org/W4308210758","https://openalex.org/W4317914880","https://openalex.org/W7160832072","https://openalex.org/W4396999785","https://openalex.org/W2919899673","https://openalex.org/W3110879213","https://openalex.org/W7124124940","https://openalex.org/W4390374156"],"abstract_inverted_index":{"Background:":[0],"3D":[1,39,128,142],"kidney":[2,23,258],"cellular":[3],"models,":[4],"organoids":[5],"or":[6,105],"tubuloids":[7,36,171,215],"that":[8],"recapitulate":[9],"tissue-specific":[10],"pathophysiology,":[11],"including":[12],"disease":[13,24,253,259,280],"phenotypes":[14],"after":[15],"genome":[16],"editing,":[17],"may":[18],"represent":[19],"useful":[20],"tools":[21],"for":[22,279],"modeling":[25],"and":[26,65,141,147,247,260],"drug":[27],"screening.":[28],"We":[29],"report":[30],"here":[31],"the":[32,91,134,174,179,187,191,208,236,264],"first":[33],"model":[34,278],"of":[35,41,77,178,194,203,210,225,266,271],"originated":[37],"from":[38,48,133,186],"culturing":[40],"human":[42,283],"collecting":[43,237],"duct":[44],"(hCD)":[45],"cells":[46,56,80,99,120,144,162,274,284],"obtained":[47],"pluripotent":[49],"stem":[50],"cells-derived":[51],"ureteric":[52],"bud":[53],"organoids.":[54],"These":[55,74],"generate":[57,95],"functional":[58,75,269],"epithelia":[59],"characterized":[60],"by":[61,151,165],"amiloride-sensitive":[62],"transepithelial":[63,67],"potential":[64],"high":[66],"resistance":[68],"when":[69],"evaluated":[70],"in":[71,90,107,110,117,160,170,173,198,223,243,282,285],"two-dimensional":[72],"transwells.":[73],"properties":[76,270],"hCD":[78,96,119,214,272],"principal":[79,273],"are":[81],"unique":[82,277],"with":[83,167,181,227,268],"no":[84],"other":[85],"comparable":[86],"cell":[87,97],"line":[88],"available":[89],"literature.":[92],"Methods:":[93],"To":[94],"tubuloids,":[98],"were":[100],"cultured":[101],"as":[102,163,250,252,256],"monolayer":[103],"(2D)":[104],"grown":[106],"Geltrex":[108],"diluted":[109],"2%":[111],"serum":[112],"DMEM/F12.":[113],"After":[114],"1-2":[115],"days":[116],"Geltrex,":[118],"formed":[121],"aggregates":[122],"which":[123,131,220],"developed":[124],"into":[125],"more":[126],"complex":[127],"tubular":[129],"structures,":[130],"detached":[132],"original":[135],"aggregates.":[136],"Results:":[137],"Both":[138],"2D":[139,161,199],"cultures":[140],"tubuloid":[143],"expressed":[145,159,216],"AVPR2":[146],"AQP2":[148,184],"gene":[149],"transcripts":[150],"qPCR.":[152],"The":[153],"water":[154],"channel,":[155],"AQP2,":[156],"was":[157],"poorly":[158],"assessed":[164],"immunofluorescence":[166],"higher":[168],"expression":[169],"primarily":[172],"intracellular":[175,188],"vesicles.":[176],"Activation":[177],"V2R":[180],"dDAVP":[182,206],"induced":[183,207],"relocation":[185],"vesicles":[189],"to":[190,205],"apical":[192],"membrane":[193],"hCD-tubuloids,":[195],"but":[196],"not":[197],"cells.":[200],"Long-term":[201],"exposure":[202],"hCD-tubuloids":[204,267],"growth":[209],"cysts.":[211],"In":[212],"addition,":[213],"PC2,":[217],"a":[218,276],"protein":[219],"is":[221],"mutated":[222],"\u02dc15%":[224],"individuals":[226],"Autosomal":[228],"Dominant":[229],"Polycystic":[230],"Kidney":[231],"Disease":[232],"(ADPKD).":[233],"Conclusions:":[234],"Since":[235],"system":[238],"plays":[239],"an":[240],"important":[241],"role":[242],"regulating":[244],"sodium,":[245],"potassium,":[246],"acid-base":[248],"homeostasis":[249],"well":[251],"states":[254],"such":[255],"polycystic":[257],"nephrogenic":[261],"diabetes":[262],"insipidus,":[263],"generation":[265],"represents":[275],"modelling":[281],"vitro.":[286],"Funding:":[287],"NIDDK":[288],"Support,":[289],"Other":[290],"NIH":[291],"Support":[292,296],"-":[293,297],"NCATs,":[294],"Government":[295],"Non-U.S.":[298]},"counts_by_year":[],"updated_date":"2026-10-16T09:59:22.235173","created_date":"2024-05-19T00:00:00"}