{"id":"https://openalex.org/W3130178042","doi":"https://doi.org/10.1080/09286586.2021.1883675","title":"The Direct Healthcare Cost of Stargardt Disease: A Claims-Based Analysis","display_name":"The Direct Healthcare Cost of Stargardt Disease: A Claims-Based Analysis","publication_year":2021,"publication_date":"2021-02-21","ids":{"openalex":"https://openalex.org/W3130178042","doi":"https://doi.org/10.1080/09286586.2021.1883675","mag":"3130178042","pmid":"https://pubmed.ncbi.nlm.nih.gov/33615979","pmcid":"https://www.ncbi.nlm.nih.gov/pmc/articles/11207193"},"language":"en","primary_location":{"id":"doi:10.1080/09286586.2021.1883675","doi":"https://doi.org/10.1080/09286586.2021.1883675","is_oa":true,"landing_page_url":"https://doi.org/10.1080/09286586.2021.1883675","pdf_url":null,"source":{"id":"https://openalex.org/S88738190","display_name":"Ophthalmic Epidemiology","issn_l":"0928-6586","issn":["0928-6586","1744-5086"],"is_oa":false,"is_in_doaj":false,"is_core":true,"listed_in":["anvur-scientific","cwts-core","dongbi-c","doyens","jufo-1","medline","norway-1","poland-70","russia-white-list-2"],"host_organization":"https://openalex.org/P4310320547","host_organization_name":"Taylor & Francis","host_organization_lineage":["https://openalex.org/P4310320547","https://openalex.org/P4310320449"],"host_organization_lineage_names":["Taylor & Francis","Informa"],"type":"journal"},"license":"cc-by-nc-nd","license_id":"https://openalex.org/licenses/cc-by-nc-nd","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Ophthalmic Epidemiology","raw_type":"journal-article"},"volume":null,"type":"article","indexed_in":["crossref","pubmed"],"open_access":{"is_oa":true,"oa_status":"hybrid","oa_url":"https://doi.org/10.1080/09286586.2021.1883675","any_repository_has_fulltext":true},"authorships":[{"author_position":"first","author":{"id":"https://openalex.org/A5055216661","display_name":"Kanza Aziz","orcid":"https://orcid.org/0000-0001-9147-9122","observed_orcids":["https://orcid.org/0000-0001-9147-9122"]},"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":"Kanza Aziz","raw_affiliation_strings":["Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA"],"raw_orcid":"https://orcid.org/0000-0001-9147-9122","affiliations":[{"raw_affiliation_string":"Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA","institution_ids":["https://openalex.org/I145311948"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5083815937","display_name":"Bonnielin K. Swenor","orcid":"https://orcid.org/0000-0002-6044-0951","observed_orcids":["https://orcid.org/0000-0002-6044-0951"]},"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":"Bonnielin K. Swenor","raw_affiliation_strings":["Department of Epidemiology, Johns Hopkins Bloomberg School of Public Health, Baltimore, Maryland, USA","Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA"],"raw_orcid":"https://orcid.org/0000-0002-6044-0951","affiliations":[{"raw_affiliation_string":"Department of Epidemiology, Johns Hopkins Bloomberg School of Public Health, Baltimore, Maryland, USA","institution_ids":["https://openalex.org/I145311948"]},{"raw_affiliation_string":"Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA","institution_ids":["https://openalex.org/I145311948"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5060417448","display_name":"Joseph K. Canner","orcid":"https://orcid.org/0000-0001-5095-8709","observed_orcids":["https://orcid.org/0000-0001-5095-8709"]},"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":"Joseph K. Canner","raw_affiliation_strings":["Johns Hopkins Surgery Center for Outcomes Research, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Johns Hopkins Surgery Center for Outcomes Research, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA","institution_ids":["https://openalex.org/I145311948","https://openalex.org/I2799853436"]}]},{"author_position":"last","author":{"id":"https://openalex.org/A5075781178","display_name":"Mandeep S. Singh","orcid":"https://orcid.org/0000-0003-1749-0088","observed_orcids":["https://orcid.org/0000-0003-1749-0088"]},"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":true,"raw_author_name":"Mandeep S. Singh","raw_affiliation_strings":["Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA"],"raw_orcid":"https://orcid.org/0000-0003-1749-0088","affiliations":[{"raw_affiliation_string":"Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA","institution_ids":["https://openalex.org/I145311948"]}]}],"institutions":[],"countries_distinct_count":1,"institutions_distinct_count":2,"corresponding_author_ids":["https://openalex.org/A5075781178"],"corresponding_institution_ids":["https://openalex.org/I145311948"],"apc_list":null,"apc_paid":null,"fwci":1.5362,"has_fulltext":false,"cited_by_count":8,"citation_normalized_percentile":{"value":0.78512677,"is_in_top_1_percent":false,"is_in_top_10_percent":false},"cited_by_percentile_year":{"min":88,"max":96},"biblio":{"volume":"28","issue":"6","first_page":"533","last_page":"539"},"is_retracted":false,"is_paratext":false,"is_xpac":false,"primary_topic":{"id":"https://openalex.org/T10170","display_name":"Retinal Diseases and Treatments","score":0.44119998812675476,"subfield":{"id":"https://openalex.org/subfields/2731","display_name":"Ophthalmology"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},"topics":[{"id":"https://openalex.org/T10170","display_name":"Retinal Diseases and Treatments","score":0.44119998812675476,"subfield":{"id":"https://openalex.org/subfields/2731","display_name":"Ophthalmology"},"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/T10691","display_name":"Retinal Development and Disorders","score":0.3833000063896179,"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/T10804","display_name":"Health Systems, Economic Evaluations, Quality of Life","score":0.0608999989926815,"subfield":{"id":"https://openalex.org/subfields/2002","display_name":"Economics and Econometrics"},"field":{"id":"https://openalex.org/fields/20","display_name":"Economics, Econometrics and Finance"},"domain":{"id":"https://openalex.org/domains/2","display_name":"Social Sciences"}}],"keywords":[{"id":"https://openalex.org/keywords/stargardt-disease","display_name":"Stargardt disease","score":1.0},{"id":"https://openalex.org/keywords/direct-healthcare-costs","display_name":"direct healthcare costs","score":0.9319999814033508},{"id":"https://openalex.org/keywords/juvenile-macular-degeneration","display_name":"juvenile macular degeneration","score":0.9160000085830688},{"id":"https://openalex.org/keywords/childhood-blindness","display_name":"childhood blindness","score":0.7059999704360962},{"id":"https://openalex.org/keywords/non-exudative-age-related-macular-degeneration","display_name":"non-exudative age-related macular degeneration","score":0.8989999890327454},{"id":"https://openalex.org/keywords/sensorineural-hearing-loss","display_name":"sensorineural hearing loss","score":0.9459999799728394},{"id":"https://openalex.org/keywords/health-insurance-claims","display_name":"health insurance claims","score":0.531000018119812},{"id":"https://openalex.org/keywords/economic-burden","display_name":"economic burden","score":0.46000000834465027},{"id":"https://openalex.org/keywords/lifetime-costs","display_name":"lifetime costs","score":0.4830000102519989}],"concepts":[{"id":"https://openalex.org/C71924100","wikidata":"https://www.wikidata.org/wiki/Q11190","display_name":"Medicine","level":0,"score":0.8958721160888672},{"id":"https://openalex.org/C2780664029","wikidata":"https://www.wikidata.org/wiki/Q5282120","display_name":"Disease burden","level":3,"score":0.5392817854881287},{"id":"https://openalex.org/C2776403814","wikidata":"https://www.wikidata.org/wiki/Q27429789","display_name":"Macular degeneration","level":2,"score":0.464387983083725},{"id":"https://openalex.org/C6964187","wikidata":"https://www.wikidata.org/wiki/Q659256","display_name":"Indirect costs","level":2,"score":0.45982125401496887},{"id":"https://openalex.org/C187212893","wikidata":"https://www.wikidata.org/wiki/Q123028","display_name":"Pediatrics","level":1,"score":0.44103696942329407},{"id":"https://openalex.org/C160735492","wikidata":"https://www.wikidata.org/wiki/Q31207","display_name":"Health care","level":2,"score":0.4206044673919678},{"id":"https://openalex.org/C149923435","wikidata":"https://www.wikidata.org/wiki/Q37732","display_name":"Demography","level":1,"score":0.33644920587539673},{"id":"https://openalex.org/C2779134260","wikidata":"https://www.wikidata.org/wiki/Q12136","display_name":"Disease","level":2,"score":0.3137270510196686},{"id":"https://openalex.org/C126322002","wikidata":"https://www.wikidata.org/wiki/Q11180","display_name":"Internal medicine","level":1,"score":0.18904882669448853},{"id":"https://openalex.org/C118487528","wikidata":"https://www.wikidata.org/wiki/Q161437","display_name":"Ophthalmology","level":1,"score":0.1536303460597992},{"id":"https://openalex.org/C144024400","wikidata":"https://www.wikidata.org/wiki/Q21201","display_name":"Sociology","level":0,"score":0.0},{"id":"https://openalex.org/C144133560","wikidata":"https://www.wikidata.org/wiki/Q4830453","display_name":"Business","level":0,"score":0.0},{"id":"https://openalex.org/C121955636","wikidata":"https://www.wikidata.org/wiki/Q4116214","display_name":"Accounting","level":1,"score":0.0},{"id":"https://openalex.org/C50522688","wikidata":"https://www.wikidata.org/wiki/Q189833","display_name":"Economic growth","level":1,"score":0.0},{"id":"https://openalex.org/C162324750","wikidata":"https://www.wikidata.org/wiki/Q8134","display_name":"Economics","level":0,"score":0.0}],"mesh":[{"descriptor_ui":"D000080362","descriptor_name":"Stargardt Disease","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D001766","descriptor_name":"Blindness","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D002648","descriptor_name":"Child","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D006801","descriptor_name":"Humans","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D016208","descriptor_name":"Databases, Factual","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D017048","descriptor_name":"Health Care Costs","qualifier_ui":null,"qualifier_name":null,"is_major_topic":true},{"descriptor_ui":"D017281","descriptor_name":"Cost of Illness","qualifier_ui":null,"qualifier_name":null,"is_major_topic":true}],"locations_count":4,"locations":[{"id":"doi:10.1080/09286586.2021.1883675","doi":"https://doi.org/10.1080/09286586.2021.1883675","is_oa":true,"landing_page_url":"https://doi.org/10.1080/09286586.2021.1883675","pdf_url":null,"source":{"id":"https://openalex.org/S88738190","display_name":"Ophthalmic Epidemiology","issn_l":"0928-6586","issn":["0928-6586","1744-5086"],"is_oa":false,"is_in_doaj":false,"is_core":true,"listed_in":["anvur-scientific","cwts-core","dongbi-c","doyens","jufo-1","medline","norway-1","poland-70","russia-white-list-2"],"host_organization":"https://openalex.org/P4310320547","host_organization_name":"Taylor & Francis","host_organization_lineage":["https://openalex.org/P4310320547","https://openalex.org/P4310320449"],"host_organization_lineage_names":["Taylor & Francis","Informa"],"type":"journal"},"license":"cc-by-nc-nd","license_id":"https://openalex.org/licenses/cc-by-nc-nd","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Ophthalmic Epidemiology","raw_type":"journal-article"},{"id":"doi:10.1101/2020.01.22.20017962","doi":"https://doi.org/10.1101/2020.01.22.20017962","is_oa":true,"landing_page_url":"https://doi.org/10.1101/2020.01.22.20017962","pdf_url":"https://www.medrxiv.org/content/medrxiv/early/2020/01/27/2020.01.22.20017962.full.pdf","source":{"id":"https://openalex.org/S3005729997","display_name":"medRxiv","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":true,"listed_in":["cwts-core"],"host_organization":null,"host_organization_name":null,"host_organization_lineage":["https://openalex.org/P4310315909"],"host_organization_lineage_names":["Cold Spring Harbor Laboratory Press"],"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":"medRxiv","raw_type":"posted-content"},{"id":"pmid:33615979","doi":null,"is_oa":false,"landing_page_url":"https://pubmed.ncbi.nlm.nih.gov/33615979","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":"Ophthalmic epidemiology","raw_type":"Journal Article"},{"id":"pmh:oai:pubmedcentral.nih.gov:11207193","doi":null,"is_oa":true,"landing_page_url":"https://www.ncbi.nlm.nih.gov/pmc/articles/11207193","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":"Ophthalmic Epidemiol","raw_type":"Text"}],"best_oa_location":{"id":"doi:10.1080/09286586.2021.1883675","doi":"https://doi.org/10.1080/09286586.2021.1883675","is_oa":true,"landing_page_url":"https://doi.org/10.1080/09286586.2021.1883675","pdf_url":null,"source":{"id":"https://openalex.org/S88738190","display_name":"Ophthalmic Epidemiology","issn_l":"0928-6586","issn":["0928-6586","1744-5086"],"is_oa":false,"is_in_doaj":false,"is_core":true,"listed_in":["anvur-scientific","cwts-core","dongbi-c","doyens","jufo-1","medline","norway-1","poland-70","russia-white-list-2"],"host_organization":"https://openalex.org/P4310320547","host_organization_name":"Taylor & Francis","host_organization_lineage":["https://openalex.org/P4310320547","https://openalex.org/P4310320449"],"host_organization_lineage_names":["Taylor & Francis","Informa"],"type":"journal"},"license":"cc-by-nc-nd","license_id":"https://openalex.org/licenses/cc-by-nc-nd","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"Ophthalmic Epidemiology","raw_type":"journal-article"},"sustainable_development_goals":[{"id":"https://metadata.un.org/sdg/3","score":0.9537000060081482,"display_name":"Good health and well-being"}],"study_designs":[{"id":"https://openalex.org/study-designs/observational-study","display_name":"Observational Study"}],"awards":[{"id":"https://openalex.org/G339113781","display_name":"Vision Loss and Cognition: Testing the sensory consequence hypothesis","funder_award_id":"K01AG052640","funder_id":"https://openalex.org/F4320337337","funder_display_name":"National Institute on Aging"},{"id":"https://openalex.org/G5806474169","display_name":null,"funder_award_id":"CD-RM-0918-0749-JHU","funder_id":"https://openalex.org/F4320306340","funder_display_name":"Foundation Fighting Blindness"}],"funders":[{"id":"https://openalex.org/F4320306340","display_name":"Foundation Fighting Blindness","ror":"https://ror.org/011728q79"},{"id":"https://openalex.org/F4320306811","display_name":"Research to Prevent Blindness","ror":"https://ror.org/04drjs621"},{"id":"https://openalex.org/F4320337337","display_name":"National Institute on Aging","ror":"https://ror.org/049v75w11"}],"has_content":{"grobid_xml":true,"pdf":true},"content_urls":{"pdf":"https://content.openalex.org/works/W3130178042.pdf","grobid_xml":"https://content.openalex.org/works/W3130178042.grobid-xml"},"referenced_works_count":24,"referenced_works":["https://openalex.org/W1939955154","https://openalex.org/W2008584269","https://openalex.org/W2016602601","https://openalex.org/W2023453045","https://openalex.org/W2037108599","https://openalex.org/W2045048112","https://openalex.org/W2056288217","https://openalex.org/W2095231843","https://openalex.org/W2102560190","https://openalex.org/W2114868925","https://openalex.org/W2118753730","https://openalex.org/W2130848396","https://openalex.org/W2139162858","https://openalex.org/W2144116593","https://openalex.org/W2149884610","https://openalex.org/W2343804072","https://openalex.org/W2416225000","https://openalex.org/W2465689492","https://openalex.org/W2505357900","https://openalex.org/W2520642007","https://openalex.org/W2594614336","https://openalex.org/W2791013442","https://openalex.org/W2888505577","https://openalex.org/W2898811160"],"related_works":["https://openalex.org/W4390126242","https://openalex.org/W1971179007","https://openalex.org/W4409479033","https://openalex.org/W2343358017","https://openalex.org/W4311106753","https://openalex.org/W1194381236","https://openalex.org/W3144507944","https://openalex.org/W7104430572","https://openalex.org/W6958555347","https://openalex.org/W2025211623"],"abstract_inverted_index":{"Abstract":[0],"Importance":[1],"Stargardt":[2],"disease":[3],"(SD)":[4],"is":[5,186],"the":[6,51,89,165],"most":[7],"common":[8],"juvenile":[9],"macular":[10,74],"degeneration":[11,75],"and":[12,22,94,123,153,176,183,216],"a":[13,30,187,221],"leading":[14],"cause":[15],"of":[16,25,40,55,60,70,114,132,145,199,213,224,238,243],"uncorrectable":[17],"childhood":[18],"blindness.":[19],"The":[20,37,128,195,233],"progressive":[21],"incurable":[23],"nature":[24],"this":[26],"chronic":[27],"condition":[28,206],"entails":[29],"long-term":[31],"financial":[32],"burden":[33],"on":[34],"affected":[35],"individuals.":[36],"economic":[38],"costs":[39],"SD":[41,136,169,200,239],"have":[42],"not":[43],"been":[44],"characterized":[45],"in":[46,211,231],"detail.":[47],"Objective":[48],"To":[49],"estimate":[50],"direct":[52,108,189],"healthcare":[53,61,109,190],"cost":[54,191,198,237],"SD.":[56,194],"Design":[57],"Cross-sectional":[58],"analysis":[59],"claims.":[62],"Participants":[63],"Patients":[64],"with":[65,193],"an":[66],"ICD-9":[67],"diagnosis":[68],"code":[69],"SD,":[71,120],"non-exudative":[72],"age-related":[73],"(AMD),":[76],"or":[77],"bilateral":[78],"sensorineural":[79],"hearing":[80],"loss":[81,226],"(SHL).":[82],"Methods":[83],"Outpatient":[84],"administrative":[85],"claims":[86],"data":[87],"from":[88,97],"IBM\u00ae":[90],"MarketScan\u00ae":[91],"Commercial":[92],"Claims":[93],"Encounters":[95],"Database":[96],"2010":[98],"to":[99],"2014":[100],"were":[101,117],"analyzed.":[102],"Main":[103],"Outcome":[104],"Measure":[105],"Annual":[106],"per-patient":[107,197,236],"cost.":[110],"Results":[111],"A":[112],"total":[113,234],"472,428":[115],"patients":[116,126],"analyzed":[118],"(5,015":[119],"369,750":[121],"SHL":[122,146,174],"97,663":[124],"AMD":[125,154,180,218],"respectively).":[127],"payment":[129,167],"per":[130],"year":[131],"insurance":[133],"coverage":[134],"for":[135,163,168],"(median:":[137,147,155],"$105.58,":[138],"IQR:":[139,149,157],"$50.53-$218.71)":[140],"was":[141,170,201],"higher":[142,172,178,202],"than":[143,173,179,203],"that":[144,207,227,242],"$51.01,":[148],"$25.66-$121.66,":[150],"p":[151,159],"<0.001)":[152],"$76.20,":[156],"$38.00-$164.86,":[158],"<0.001).":[160],"When":[161],"adjusted":[162],"covariates,":[164],"annual":[166,196],"$47.83":[171],"(p<0.001)":[175],"$17.34":[177],"(p<0.001).":[181],"Conclusions":[182],"Relevance":[184],"There":[185],"significant":[188],"associated":[192],"SHL,":[204],"another":[205],"causes":[208,220],"sensory":[209],"impairment":[210],"people":[212],"all":[214],"ages,":[215],"nonexudative":[217,244],"which":[219],"similar":[222],"pattern":[223],"visual":[225],"typically":[228],"begins":[229],"later":[230],"life.":[232],"lifetime":[235],"may":[240],"exceed":[241],"AMD.":[245]},"counts_by_year":[{"year":2025,"cited_by_count":1},{"year":2024,"cited_by_count":3},{"year":2023,"cited_by_count":2},{"year":2022,"cited_by_count":1},{"year":2021,"cited_by_count":1}],"updated_date":"2026-11-04T10:54:05.112534","created_date":"2021-03-01T00:00:00"}
