{"id":"https://openalex.org/W2032776853","doi":"https://doi.org/10.1074/jbc.m600807200","title":"Lysosomal Localization of TRPML3 Depends on TRPML2 and the Mucolipidosis-associated Protein TRPML1","display_name":"Lysosomal Localization of TRPML3 Depends on TRPML2 and the Mucolipidosis-associated Protein TRPML1","publication_year":2006,"publication_date":"2006-04-11","ids":{"openalex":"https://openalex.org/W2032776853","doi":"https://doi.org/10.1074/jbc.m600807200","mag":"2032776853","pmid":"https://pubmed.ncbi.nlm.nih.gov/16606612","pmcid":"https://www.ncbi.nlm.nih.gov/pmc/articles/4196876"},"language":"en","primary_location":{"id":"doi:10.1074/jbc.m600807200","doi":"https://doi.org/10.1074/jbc.m600807200","is_oa":true,"landing_page_url":"https://doi.org/10.1074/jbc.m600807200","pdf_url":"http://www.jbc.org/article/S0021925820557815/pdf","source":{"id":"https://openalex.org/S140251998","display_name":"Journal of Biological Chemistry","issn_l":"0021-9258","issn":["0021-9258","1067-8816","1083-351X"],"is_oa":false,"is_in_doaj":true,"is_core":true,"listed_in":["anvur-scientific","cwts-core","doaj","dongbi-b","doyens","jufo-2","ki-jl-2","medline","norway-2","poland-100","vabb-shw"],"host_organization":"https://openalex.org/P4310320990","host_organization_name":"Elsevier BV","host_organization_lineage":["https://openalex.org/P4310320990"],"host_organization_lineage_names":["Elsevier BV"],"type":"journal"},"license":"cc-by","license_id":"https://openalex.org/licenses/cc-by","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Journal of Biological Chemistry","raw_type":"journal-article"},"volume":null,"type":"article","indexed_in":["crossref","doaj","pubmed"],"open_access":{"is_oa":true,"oa_status":"hybrid","oa_url":"http://www.jbc.org/article/S0021925820557815/pdf","any_repository_has_fulltext":true},"authorships":[{"author_position":"first","author":{"id":"https://openalex.org/A5017438792","display_name":"Kartik Venkatachalam","orcid":"https://orcid.org/0000-0002-3055-9265","observed_orcids":["https://orcid.org/0000-0002-3055-9265"]},"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"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Kartik Venkatachalam","raw_affiliation_strings":["Department of Biological Chemistry, The Johns Hopkins University School of Medicine, Baltimore, Maryland 21205"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Biological Chemistry, The Johns Hopkins University School of Medicine, Baltimore, Maryland 21205","institution_ids":["https://openalex.org/I145311948"]}]},{"author_position":"middle","author":{"id":null,"display_name":"Thomas Hofmann","orcid":null,"observed_orcids":[]},"institutions":[{"id":"https://openalex.org/I161103922","display_name":"Philipps University of Marburg","ror":"https://ror.org/01rdrb571","country_code":"DE","type":"education","lineage":["https://openalex.org/I161103922"]}],"countries":["DE"],"is_corresponding":false,"raw_author_name":"Thomas Hofmann","raw_affiliation_strings":["Institut fuer Pharmakologie und Toxikologie, Philipps-Universitaet Marburg, Karl-von-Frisch-Strasse 1, 35033 Marburg, Germany"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Institut fuer Pharmakologie und Toxikologie, Philipps-Universitaet Marburg, Karl-von-Frisch-Strasse 1, 35033 Marburg, Germany","institution_ids":["https://openalex.org/I161103922"]}]},{"author_position":"last","author":{"id":"https://openalex.org/A5059045594","display_name":"Craig Montell","orcid":"https://orcid.org/0000-0001-5637-1482","observed_orcids":["https://orcid.org/0000-0001-5637-1482"]},"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":"Craig Montell","raw_affiliation_strings":["Department of Biological Chemistry, The Johns Hopkins University School of Medicine, Baltimore, Maryland 21205. Electronic address: cmontell@jhmi.edu","Department of Biological Chemistry The Johns Hopkins University School of Medicine Baltimore, Maryland 21205"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Biological Chemistry, The Johns Hopkins University School of Medicine, Baltimore, Maryland 21205. Electronic address: cmontell@jhmi.edu","institution_ids":["https://openalex.org/I145311948","https://openalex.org/I2799853436"]},{"raw_affiliation_string":"Department of Biological Chemistry The Johns Hopkins University School of Medicine Baltimore, Maryland 21205","institution_ids":["https://openalex.org/I145311948"]}]}],"institutions":[],"countries_distinct_count":2,"institutions_distinct_count":3,"corresponding_author_ids":["https://openalex.org/A5059045594"],"corresponding_institution_ids":["https://openalex.org/I145311948","https://openalex.org/I2799853436"],"apc_list":{"value":3430,"currency":"USD","value_usd":3430},"apc_paid":{"value":3430,"currency":"USD","value_usd":3430},"fwci":7.5538,"has_fulltext":true,"cited_by_count":149,"citation_normalized_percentile":{"value":0.98698482,"is_in_top_1_percent":false,"is_in_top_10_percent":true},"cited_by_percentile_year":{"min":94,"max":99},"biblio":{"volume":"281","issue":"25","first_page":"17517","last_page":"17527"},"is_retracted":false,"is_paratext":false,"is_xpac":false,"primary_topic":{"id":"https://openalex.org/T13415","display_name":"Calcium signaling and nucleotide metabolism","score":0.9984999895095825,"subfield":{"id":"https://openalex.org/subfields/1314","display_name":"Physiology"},"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/T13415","display_name":"Calcium signaling and nucleotide metabolism","score":0.9984999895095825,"subfield":{"id":"https://openalex.org/subfields/1314","display_name":"Physiology"},"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/T10617","display_name":"Cellular transport and secretion","score":0.0005000000237487257,"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"}},{"id":"https://openalex.org/T11204","display_name":"Ion Channels and Receptors","score":0.0005000000237487257,"subfield":{"id":"https://openalex.org/subfields/2809","display_name":"Sensory Systems"},"field":{"id":"https://openalex.org/fields/28","display_name":"Neuroscience"},"domain":{"id":"https://openalex.org/domains/1","display_name":"Life Sciences"}}],"keywords":[{"id":"https://openalex.org/keywords/trpml3","display_name":"TRPML3","score":0.9589999914169312},{"id":"https://openalex.org/keywords/trpml1","display_name":"TRPML1","score":0.9169999957084656},{"id":"https://openalex.org/keywords/trpml2","display_name":"TRPML2","score":1.0},{"id":"https://openalex.org/keywords/mucolipidosis-type-iv","display_name":"mucolipidosis type IV","score":0.9440000057220459},{"id":"https://openalex.org/keywords/lysosomal-storage-disorders","display_name":"lysosomal storage disease","score":0.7390000224113464},{"id":"https://openalex.org/keywords/lysosomal-localization","display_name":"lysosomal localization","score":0.7900000214576721},{"id":"https://openalex.org/keywords/protein-oligomerization","display_name":"protein oligomerization","score":0.7789999842643738},{"id":"https://openalex.org/keywords/protein-trafficking","display_name":"protein trafficking","score":0.4860000014305115},{"id":"https://openalex.org/keywords/er-retention","display_name":"endoplasmic reticulum retention","score":0.8420000076293945},{"id":"https://openalex.org/keywords/subcellular-localization","display_name":"subcellular localization","score":0.7139999866485596},{"id":"https://openalex.org/keywords/cation-channels","display_name":"cation channels","score":0.4449999928474426},{"id":"https://openalex.org/keywords/corneal-opacity","display_name":"corneal opacity","score":0.7350000143051147}],"concepts":[{"id":"https://openalex.org/C158617107","wikidata":"https://www.wikidata.org/wiki/Q79927","display_name":"Endoplasmic reticulum","level":2,"score":0.792397141456604},{"id":"https://openalex.org/C95444343","wikidata":"https://www.wikidata.org/wiki/Q7141","display_name":"Cell biology","level":1,"score":0.6339915990829468},{"id":"https://openalex.org/C2776925932","wikidata":"https://www.wikidata.org/wiki/Q1755122","display_name":"Neurodegeneration","level":3,"score":0.6008170247077942},{"id":"https://openalex.org/C2778264360","wikidata":"https://www.wikidata.org/wiki/Q675010","display_name":"Lysosomal storage disease","level":3,"score":0.5877125263214111},{"id":"https://openalex.org/C2780216420","wikidata":"https://www.wikidata.org/wiki/Q83330","display_name":"Lysosome","level":3,"score":0.5863357186317444},{"id":"https://openalex.org/C86803240","wikidata":"https://www.wikidata.org/wiki/Q420","display_name":"Biology","level":0,"score":0.5838122367858887},{"id":"https://openalex.org/C155164980","wikidata":"https://www.wikidata.org/wiki/Q304043","display_name":"Transient receptor potential channel","level":3,"score":0.4965711236000061},{"id":"https://openalex.org/C2776879804","wikidata":"https://www.wikidata.org/wiki/Q7630919","display_name":"Subcellular localization","level":3,"score":0.4795917570590973},{"id":"https://openalex.org/C2780905385","wikidata":"https://www.wikidata.org/wiki/Q1952032","display_name":"Mucolipidosis","level":3,"score":0.45181044936180115},{"id":"https://openalex.org/C55493867","wikidata":"https://www.wikidata.org/wiki/Q7094","display_name":"Biochemistry","level":1,"score":0.3272216022014618},{"id":"https://openalex.org/C170493617","wikidata":"https://www.wikidata.org/wiki/Q208467","display_name":"Receptor","level":2,"score":0.1653347611427307},{"id":"https://openalex.org/C190062978","wikidata":"https://www.wikidata.org/wiki/Q79899","display_name":"Cytoplasm","level":2,"score":0.14824062585830688},{"id":"https://openalex.org/C126322002","wikidata":"https://www.wikidata.org/wiki/Q11180","display_name":"Internal medicine","level":1,"score":0.11554029583930969},{"id":"https://openalex.org/C181199279","wikidata":"https://www.wikidata.org/wiki/Q8047","display_name":"Enzyme","level":2,"score":0.07749989628791809},{"id":"https://openalex.org/C71924100","wikidata":"https://www.wikidata.org/wiki/Q11190","display_name":"Medicine","level":0,"score":0.0},{"id":"https://openalex.org/C2779134260","wikidata":"https://www.wikidata.org/wiki/Q12136","display_name":"Disease","level":2,"score":0.0}],"mesh":[{"descriptor_ui":"D000818","descriptor_name":"Animals","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D002460","descriptor_name":"Cell Line","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D002462","descriptor_name":"Cell Membrane","qualifier_ui":"Q000378","qualifier_name":"metabolism","is_major_topic":false},{"descriptor_ui":"D006801","descriptor_name":"Humans","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D008247","descriptor_name":"Lysosomes","qualifier_ui":"Q000378","qualifier_name":"metabolism","is_major_topic":true},{"descriptor_ui":"D009081","descriptor_name":"Mucolipidoses","qualifier_ui":"Q000378","qualifier_name":"metabolism","is_major_topic":false},{"descriptor_ui":"D041681","descriptor_name":"NIH 3T3 Cells","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D050051","descriptor_name":"Transient Receptor Potential Channels","qualifier_ui":"Q000096","qualifier_name":"biosynthesis","is_major_topic":true},{"descriptor_ui":"D050051","descriptor_name":"Transient Receptor Potential Channels","qualifier_ui":"Q000502","qualifier_name":"physiology","is_major_topic":true},{"descriptor_ui":"D050053","descriptor_name":"TRPM Cation Channels","qualifier_ui":"Q000096","qualifier_name":"biosynthesis","is_major_topic":true},{"descriptor_ui":"D050053","descriptor_name":"TRPM Cation Channels","qualifier_ui":"Q000502","qualifier_name":"physiology","is_major_topic":true},{"descriptor_ui":"D051379","descriptor_name":"Mice","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false}],"locations_count":3,"locations":[{"id":"doi:10.1074/jbc.m600807200","doi":"https://doi.org/10.1074/jbc.m600807200","is_oa":true,"landing_page_url":"https://doi.org/10.1074/jbc.m600807200","pdf_url":"http://www.jbc.org/article/S0021925820557815/pdf","source":{"id":"https://openalex.org/S140251998","display_name":"Journal of Biological Chemistry","issn_l":"0021-9258","issn":["0021-9258","1067-8816","1083-351X"],"is_oa":false,"is_in_doaj":true,"is_core":true,"listed_in":["anvur-scientific","cwts-core","doaj","dongbi-b","doyens","jufo-2","ki-jl-2","medline","norway-2","poland-100","vabb-shw"],"host_organization":"https://openalex.org/P4310320990","host_organization_name":"Elsevier BV","host_organization_lineage":["https://openalex.org/P4310320990"],"host_organization_lineage_names":["Elsevier BV"],"type":"journal"},"license":"cc-by","license_id":"https://openalex.org/licenses/cc-by","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Journal of Biological Chemistry","raw_type":"journal-article"},{"id":"pmid:16606612","doi":null,"is_oa":false,"landing_page_url":"https://pubmed.ncbi.nlm.nih.gov/16606612","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 biological chemistry","raw_type":"Journal Article"},{"id":"pmh:oai:pubmedcentral.nih.gov:4196876","doi":null,"is_oa":true,"landing_page_url":"https://www.ncbi.nlm.nih.gov/pmc/articles/4196876","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":"acceptedVersion","is_accepted":true,"is_published":false,"raw_source_name":"J Biol Chem","raw_type":"Text"}],"best_oa_location":{"id":"doi:10.1074/jbc.m600807200","doi":"https://doi.org/10.1074/jbc.m600807200","is_oa":true,"landing_page_url":"https://doi.org/10.1074/jbc.m600807200","pdf_url":"http://www.jbc.org/article/S0021925820557815/pdf","source":{"id":"https://openalex.org/S140251998","display_name":"Journal of Biological Chemistry","issn_l":"0021-9258","issn":["0021-9258","1067-8816","1083-351X"],"is_oa":false,"is_in_doaj":true,"is_core":true,"listed_in":["anvur-scientific","cwts-core","doaj","dongbi-b","doyens","jufo-2","ki-jl-2","medline","norway-2","poland-100","vabb-shw"],"host_organization":"https://openalex.org/P4310320990","host_organization_name":"Elsevier BV","host_organization_lineage":["https://openalex.org/P4310320990"],"host_organization_lineage_names":["Elsevier BV"],"type":"journal"},"license":"cc-by","license_id":"https://openalex.org/licenses/cc-by","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Journal of Biological Chemistry","raw_type":"journal-article"},"sustainable_development_goals":[{"id":"https://metadata.un.org/sdg/3","score":0.5683000087738037,"display_name":"Good health and well-being"}],"study_designs":[],"awards":[{"id":"https://openalex.org/G2171598571","display_name":null,"funder_award_id":"EY10852","funder_id":"https://openalex.org/F4320337350","funder_display_name":"National Eye Institute"},{"id":"https://openalex.org/G2818762616","display_name":"Regulation of TRP channels and visual transduction","funder_award_id":"R01EY010852","funder_id":"https://openalex.org/F4320337350","funder_display_name":"National Eye Institute"}],"funders":[{"id":"https://openalex.org/F4320310205","display_name":"Mucolipidosis IV Foundation","ror":"https://ror.org/01ajpw658"},{"id":"https://openalex.org/F4320332161","display_name":"National Institutes of Health","ror":"https://ror.org/01cwqze88"},{"id":"https://openalex.org/F4320337350","display_name":"National Eye Institute","ror":"https://ror.org/03wkg3b53"}],"has_content":{"grobid_xml":true,"pdf":true},"content_urls":{"pdf":"https://content.openalex.org/works/W2032776853.pdf","grobid_xml":"https://content.openalex.org/works/W2032776853.grobid-xml"},"referenced_works_count":43,"referenced_works":["https://openalex.org/W57374400","https://openalex.org/W1510679795","https://openalex.org/W1510849499","https://openalex.org/W1511506036","https://openalex.org/W1595083713","https://openalex.org/W1698471795","https://openalex.org/W1963669764","https://openalex.org/W1965271485","https://openalex.org/W1968704909","https://openalex.org/W1971826341","https://openalex.org/W1976514154","https://openalex.org/W1995692886","https://openalex.org/W1996149118","https://openalex.org/W1996764186","https://openalex.org/W1997112574","https://openalex.org/W2000846031","https://openalex.org/W2008303354","https://openalex.org/W2011625746","https://openalex.org/W2013180604","https://openalex.org/W2015272827","https://openalex.org/W2016354937","https://openalex.org/W2017011727","https://openalex.org/W2026062772","https://openalex.org/W2033808333","https://openalex.org/W2040987875","https://openalex.org/W2055385755","https://openalex.org/W2060799319","https://openalex.org/W2063186078","https://openalex.org/W2081434136","https://openalex.org/W2102204826","https://openalex.org/W2106744303","https://openalex.org/W2108629058","https://openalex.org/W2124512829","https://openalex.org/W2131824360","https://openalex.org/W2133285107","https://openalex.org/W2148351021","https://openalex.org/W2150091977","https://openalex.org/W2151512622","https://openalex.org/W2159244376","https://openalex.org/W2160913295","https://openalex.org/W2161518588","https://openalex.org/W2165290124","https://openalex.org/W2167061320"],"related_works":["https://openalex.org/W1976527854","https://openalex.org/W2120965833","https://openalex.org/W125928855","https://openalex.org/W2093939917","https://openalex.org/W4250191449","https://openalex.org/W2516137352","https://openalex.org/W2059052368","https://openalex.org/W4251027046","https://openalex.org/W2794016096","https://openalex.org/W2037727418"],"abstract_inverted_index":{"Mucolipidosis":[0],"type":[1],"IV":[2],"is":[3,125,135,156],"an":[4],"autosomal":[5],"recessive":[6],"lysosomal":[7,102,129],"storage":[8],"disorder":[9],"characterized":[10],"by":[11],"severe":[12],"neurodegeneration,":[13],"achlorhydria,":[14],"and":[15,22,49,63,80,98,124,133,169,176],"visual":[16],"impairments":[17],"such":[18,166],"as":[19],"corneal":[20],"opacity":[21],"strabismus.":[23],"The":[24],"disease":[25],"arises":[26],"due":[27],"to":[28,58,77,115],"mutations":[29],"in":[30,108,147],"a":[31,157],"group":[32],"2":[33],"transient":[34],"receptor":[35],"potential":[36],"(TRP)-related":[37],"cation":[38],"channel,":[39],"TRPML1.":[40],"Mammals":[41],"encode":[42],"two":[43],"additional":[44],"TRPML":[45],"proteins":[46,57],"named":[47],"TRPML2":[48,89,99,123,134,146,170],"TRPML3.":[50,96],"Information":[51],"regarding":[52],"the":[53,83,92,109,148,160,164,172],"propensity":[54],"of":[55,85,95,131,143,163,174],"these":[56],"multimerize,":[59],"their":[60,67],"subcellular":[61,161],"distribution":[62,94],"mechanisms":[64],"that":[65,74,154,167],"regulate":[66],"trafficking":[68],"are":[69,101,107],"limited.":[70],"Here":[71],"we":[72],"demonstrate":[73,153],"TRPMLs":[75,165],"interact":[76],"form":[78],"homo-":[79],"heteromultimers.":[81],"Moreover,":[82],"presence":[84],"either":[86,120],"TRPML1":[87,97,121,132,144,168],"or":[88,122,145],"specifically":[90],"influences":[91],"spatial":[93],"homomultimers":[100,106],"proteins,":[103],"whereas":[104],"TRPML3":[105,113,138,175],"endoplasmic":[110,149],"reticulum.":[111,150],"However,":[112],"localizes":[114],"lysosomes":[116],"when":[117,128],"coexpressed":[118],"with":[119],"comparably":[126],"mislocalized":[127],"targeting":[130],"disrupted.":[136],"Conversely,":[137],"does":[139],"not":[140,177],"cause":[141],"retention":[142],"These":[151],"data":[152],"there":[155],"hierarchy":[158],"controlling":[159],"distributions":[162],"dictate":[171],"localization":[173],"vice":[178],"versa.":[179]},"counts_by_year":[{"year":2026,"cited_by_count":2},{"year":2025,"cited_by_count":3},{"year":2024,"cited_by_count":2},{"year":2023,"cited_by_count":7},{"year":2022,"cited_by_count":8},{"year":2021,"cited_by_count":6},{"year":2020,"cited_by_count":7},{"year":2019,"cited_by_count":5},{"year":2018,"cited_by_count":4},{"year":2017,"cited_by_count":7},{"year":2016,"cited_by_count":3},{"year":2015,"cited_by_count":8},{"year":2014,"cited_by_count":9},{"year":2013,"cited_by_count":9},{"year":2012,"cited_by_count":12}],"updated_date":"2026-10-31T12:09:11.096373","created_date":"2016-06-24T00:00:00"}