{"id":"https://openalex.org/W4393900528","doi":"https://doi.org/10.1002/jha2.882","title":"Systematic review and evidence gap assessment of the clinical, quality of life, and economic burden of alpha\u2010thalassemia","display_name":"Systematic review and evidence gap assessment of the clinical, quality of life, and economic burden of alpha\u2010thalassemia","publication_year":2024,"publication_date":"2024-04-02","ids":{"openalex":"https://openalex.org/W4393900528","doi":"https://doi.org/10.1002/jha2.882","pmid":"https://pubmed.ncbi.nlm.nih.gov/38895066","pmcid":"https://www.ncbi.nlm.nih.gov/pmc/articles/11182398"},"language":"en","primary_location":{"id":"doi:10.1002/jha2.882","doi":"https://doi.org/10.1002/jha2.882","is_oa":true,"landing_page_url":"https://doi.org/10.1002/jha2.882","pdf_url":"https://onlinelibrary.wiley.com/doi/pdfdirect/10.1002/jha2.882","source":{"id":"https://openalex.org/S4210227740","display_name":"eJHaem","issn_l":"2688-6146","issn":["2688-6146"],"is_oa":true,"is_in_doaj":true,"is_core":true,"listed_in":["cwts-core","doaj","jufo-1","ki-jl-1","norway-1","oarl"],"host_organization":"https://openalex.org/P4310320595","host_organization_name":"Wiley","host_organization_lineage":["https://openalex.org/P4310320595"],"host_organization_lineage_names":["Wiley"],"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":"eJHaem","raw_type":"journal-article"},"volume":null,"type":"review","indexed_in":["crossref","doaj","pubmed"],"open_access":{"is_oa":true,"oa_status":"gold","oa_url":"https://onlinelibrary.wiley.com/doi/pdfdirect/10.1002/jha2.882","any_repository_has_fulltext":true},"authorships":[{"author_position":"first","author":{"id":"https://openalex.org/A5055062993","display_name":"Khaled M. Musallam","orcid":"https://orcid.org/0000-0003-3935-903X","observed_orcids":["https://orcid.org/0000-0003-3935-903X"]},"institutions":[],"countries":[],"is_corresponding":true,"raw_author_name":"Khaled M. Musallam","raw_affiliation_strings":["Center for Research on Rare Blood Disorders (CR-RBD), Burjeel Medical City, Abu Dhabi, United Arab Emirates"],"raw_orcid":"https://orcid.org/0000-0003-3935-903X","affiliations":[{"raw_affiliation_string":"Center for Research on Rare Blood Disorders (CR-RBD), Burjeel Medical City, Abu Dhabi, United Arab Emirates","institution_ids":[]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5080150203","display_name":"Vip Viprakasit","orcid":"https://orcid.org/0000-0003-3162-1849","observed_orcids":["https://orcid.org/0000-0003-3162-1849"]},"institutions":[{"id":"https://openalex.org/I1332387789","display_name":"Siriraj Hospital","ror":"https://ror.org/0331zs648","country_code":"TH","type":"healthcare","lineage":["https://openalex.org/I1332387789"]},{"id":"https://openalex.org/I25399158","display_name":"Mahidol University","ror":"https://ror.org/01znkr924","country_code":"TH","type":"education","lineage":["https://openalex.org/I25399158"]}],"countries":["TH"],"is_corresponding":false,"raw_author_name":"Vip Viprakasit","raw_affiliation_strings":["Department of Pediatrics &amp; Thalassemia Center Faculty of Medicine Siriraj Hospital Mahidol University  Bangkok Thailand","Department of Pediatrics & Thalassemia Center, Faculty of Medicine, Siriraj Hospital, Mahidol University, Bangkok, Thailand"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Pediatrics &amp; Thalassemia Center Faculty of Medicine Siriraj Hospital Mahidol University  Bangkok Thailand","institution_ids":["https://openalex.org/I1332387789","https://openalex.org/I25399158"]},{"raw_affiliation_string":"Department of Pediatrics & Thalassemia Center, Faculty of Medicine, Siriraj Hospital, Mahidol University, Bangkok, Thailand","institution_ids":["https://openalex.org/I1332387789","https://openalex.org/I25399158"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5047285892","display_name":"Louise Lombard","orcid":null,"observed_orcids":[]},"institutions":[{"id":"https://openalex.org/I4210092565","display_name":"Agios Pharmaceuticals (United States)","ror":"https://ror.org/002x06r10","country_code":"US","type":"company","lineage":["https://openalex.org/I4210092565"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Louise Lombard","raw_affiliation_strings":["Agios Pharmaceuticals, Cambridge, Massachusetts, USA"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Agios Pharmaceuticals, Cambridge, Massachusetts, USA","institution_ids":["https://openalex.org/I4210092565"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5066227176","display_name":"Keely Gilroy","orcid":null,"observed_orcids":[]},"institutions":[{"id":"https://openalex.org/I4210092565","display_name":"Agios Pharmaceuticals (United States)","ror":"https://ror.org/002x06r10","country_code":"US","type":"company","lineage":["https://openalex.org/I4210092565"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Keely Gilroy","raw_affiliation_strings":["Agios Pharmaceuticals, Cambridge, Massachusetts, USA"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Agios Pharmaceuticals, Cambridge, Massachusetts, USA","institution_ids":["https://openalex.org/I4210092565"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5109632879","display_name":"Amey Rane","orcid":null,"observed_orcids":[]},"institutions":[{"id":"https://openalex.org/I4210092565","display_name":"Agios Pharmaceuticals (United States)","ror":"https://ror.org/002x06r10","country_code":"US","type":"company","lineage":["https://openalex.org/I4210092565"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Amey Rane","raw_affiliation_strings":["Agios Pharmaceuticals, Cambridge, Massachusetts, USA"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Agios Pharmaceuticals, Cambridge, Massachusetts, USA","institution_ids":["https://openalex.org/I4210092565"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5042062992","display_name":"Lydia Vinals","orcid":"https://orcid.org/0009-0003-8465-3358","observed_orcids":["https://orcid.org/0009-0003-8465-3358"]},"institutions":[],"countries":[],"is_corresponding":false,"raw_author_name":"Lydia Vinals","raw_affiliation_strings":["Cytel, Toronto, Ontario, Canada"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Cytel, Toronto, Ontario, Canada","institution_ids":[]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5042540832","display_name":"Candice Tam","orcid":null,"observed_orcids":[]},"institutions":[],"countries":[],"is_corresponding":false,"raw_author_name":"Candice Tam","raw_affiliation_strings":["Cytel, London, UK"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Cytel, London, UK","institution_ids":[]}]},{"author_position":"middle","author":{"id":null,"display_name":"Maria Rizzo","orcid":null,"observed_orcids":[]},"institutions":[],"countries":[],"is_corresponding":false,"raw_author_name":"Maria Rizzo","raw_affiliation_strings":["Cytel, London, UK"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Cytel, London, UK","institution_ids":[]}]},{"author_position":"last","author":{"id":"https://openalex.org/A5077729374","display_name":"Thomas D. Coates","orcid":"https://orcid.org/0000-0001-9878-6029","observed_orcids":["https://orcid.org/0000-0001-9878-6029"]},"institutions":[{"id":"https://openalex.org/I1174212","display_name":"University of Southern California","ror":"https://ror.org/03taz7m60","country_code":"US","type":"education","lineage":["https://openalex.org/I1174212"]},{"id":"https://openalex.org/I1336910626","display_name":"Children's Hospital Los Angeles","ror":"https://ror.org/00412ts95","country_code":"US","type":"healthcare","lineage":["https://openalex.org/I1336910626"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"Thomas D. Coates","raw_affiliation_strings":["Cancer and Blood Disease Institute, Children's Hospital Los Angeles and USC Keck School of Medicine, Los Angeles, California, USA"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Cancer and Blood Disease Institute, Children's Hospital Los Angeles and USC Keck School of Medicine, Los Angeles, California, USA","institution_ids":["https://openalex.org/I1174212","https://openalex.org/I1336910626"]}]}],"institutions":[],"countries_distinct_count":2,"institutions_distinct_count":5,"corresponding_author_ids":["https://openalex.org/A5055062993"],"corresponding_institution_ids":[],"apc_list":{"value":2600,"currency":"USD","value_usd":2600},"apc_paid":{"value":2600,"currency":"USD","value_usd":2600},"fwci":0.8326,"has_fulltext":true,"cited_by_count":11,"citation_normalized_percentile":{"value":0.61428571,"is_in_top_1_percent":false,"is_in_top_10_percent":false},"cited_by_percentile_year":{"min":96,"max":99},"biblio":{"volume":"5","issue":"3","first_page":"541","last_page":"547"},"is_retracted":false,"is_paratext":false,"is_xpac":false,"primary_topic":{"id":"https://openalex.org/T10554","display_name":"Hemoglobinopathies and Related Disorders","score":1.0,"subfield":{"id":"https://openalex.org/subfields/2716","display_name":"Genetics"},"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/T10554","display_name":"Hemoglobinopathies and Related Disorders","score":1.0,"subfield":{"id":"https://openalex.org/subfields/2716","display_name":"Genetics"},"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/T10804","display_name":"Health Systems, Economic Evaluations, Quality of Life","score":0.0,"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"}},{"id":"https://openalex.org/T10452","display_name":"Iron Metabolism and Disorders","score":0.0,"subfield":{"id":"https://openalex.org/subfields/2720","display_name":"Hematology"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}}],"keywords":[{"id":"https://openalex.org/keywords/alpha-thalassemia","display_name":"alpha-thalassemia","score":0.9950000047683716},{"id":"https://openalex.org/keywords/health-related-quality-of-life","display_name":"health-related quality of life","score":0.8730000257492065},{"id":"https://openalex.org/keywords/economic-burden","display_name":"economic burden","score":0.9860000014305115},{"id":"https://openalex.org/keywords/clinical-burden","display_name":"clinical burden","score":0.8600000143051147},{"id":"https://openalex.org/keywords/evidence-gaps","display_name":"evidence gaps","score":0.734000027179718},{"id":"https://openalex.org/keywords/morbidity","display_name":"morbidity","score":0.6669999957084656}],"concepts":[{"id":"https://openalex.org/C189708586","wikidata":"https://www.wikidata.org/wiki/Q1504425","display_name":"Systematic review","level":3,"score":0.7041807770729065},{"id":"https://openalex.org/C2777799968","wikidata":"https://www.wikidata.org/wiki/Q185137","display_name":"Thalassemia","level":2,"score":0.6270412802696228},{"id":"https://openalex.org/C71924100","wikidata":"https://www.wikidata.org/wiki/Q11190","display_name":"Medicine","level":0,"score":0.6197956204414368},{"id":"https://openalex.org/C2779951463","wikidata":"https://www.wikidata.org/wiki/Q7268788","display_name":"Quality of life (healthcare)","level":2,"score":0.523207426071167},{"id":"https://openalex.org/C177713679","wikidata":"https://www.wikidata.org/wiki/Q679690","display_name":"Intensive care medicine","level":1,"score":0.45071280002593994},{"id":"https://openalex.org/C2779231881","wikidata":"https://www.wikidata.org/wiki/Q5977147","display_name":"Medical literature","level":2,"score":0.43959206342697144},{"id":"https://openalex.org/C2779473830","wikidata":"https://www.wikidata.org/wiki/Q1540899","display_name":"MEDLINE","level":2,"score":0.3910055160522461},{"id":"https://openalex.org/C142724271","wikidata":"https://www.wikidata.org/wiki/Q7208","display_name":"Pathology","level":1,"score":0.08488750457763672},{"id":"https://openalex.org/C17744445","wikidata":"https://www.wikidata.org/wiki/Q36442","display_name":"Political science","level":0,"score":0.08381333947181702},{"id":"https://openalex.org/C126322002","wikidata":"https://www.wikidata.org/wiki/Q11180","display_name":"Internal medicine","level":1,"score":0.05924054980278015},{"id":"https://openalex.org/C159110408","wikidata":"https://www.wikidata.org/wiki/Q121176","display_name":"Nursing","level":1,"score":0.05454361438751221},{"id":"https://openalex.org/C199539241","wikidata":"https://www.wikidata.org/wiki/Q7748","display_name":"Law","level":1,"score":0.0}],"mesh":[],"locations_count":4,"locations":[{"id":"doi:10.1002/jha2.882","doi":"https://doi.org/10.1002/jha2.882","is_oa":true,"landing_page_url":"https://doi.org/10.1002/jha2.882","pdf_url":"https://onlinelibrary.wiley.com/doi/pdfdirect/10.1002/jha2.882","source":{"id":"https://openalex.org/S4210227740","display_name":"eJHaem","issn_l":"2688-6146","issn":["2688-6146"],"is_oa":true,"is_in_doaj":true,"is_core":true,"listed_in":["cwts-core","doaj","jufo-1","ki-jl-1","norway-1","oarl"],"host_organization":"https://openalex.org/P4310320595","host_organization_name":"Wiley","host_organization_lineage":["https://openalex.org/P4310320595"],"host_organization_lineage_names":["Wiley"],"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":"eJHaem","raw_type":"journal-article"},{"id":"pmh:oai:pubmedcentral.nih.gov:11182398","doi":null,"is_oa":true,"landing_page_url":"https://www.ncbi.nlm.nih.gov/pmc/articles/11182398","pdf_url":"https://pmc.ncbi.nlm.nih.gov/articles/PMC11182398/pdf/JHA2-5-541.pdf","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":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"EJHaem","raw_type":"Text"},{"id":"pmid:38895066","doi":null,"is_oa":false,"landing_page_url":"https://pubmed.ncbi.nlm.nih.gov/38895066","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":"EJHaem","raw_type":"Journal Article"},{"id":"pmh:oai:doaj.org/article:623abbb4023745b99d12f3d38593f588","doi":null,"is_oa":true,"landing_page_url":"https://doaj.org/article/623abbb4023745b99d12f3d38593f588","pdf_url":null,"source":{"id":"https://openalex.org/S4306401280","display_name":"DOAJ (DOAJ: Directory of Open Access Journals)","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":null,"host_organization_name":null,"host_organization_lineage":[],"host_organization_lineage_names":[],"type":"repository"},"license":"cc-by-sa","license_id":"https://openalex.org/licenses/cc-by-sa","version":"submittedVersion","is_accepted":false,"is_published":false,"raw_source_name":"eJHaem, Vol 5, Iss 3, Pp 541-547 (2024)","raw_type":"article"}],"best_oa_location":{"id":"doi:10.1002/jha2.882","doi":"https://doi.org/10.1002/jha2.882","is_oa":true,"landing_page_url":"https://doi.org/10.1002/jha2.882","pdf_url":"https://onlinelibrary.wiley.com/doi/pdfdirect/10.1002/jha2.882","source":{"id":"https://openalex.org/S4210227740","display_name":"eJHaem","issn_l":"2688-6146","issn":["2688-6146"],"is_oa":true,"is_in_doaj":true,"is_core":true,"listed_in":["cwts-core","doaj","jufo-1","ki-jl-1","norway-1","oarl"],"host_organization":"https://openalex.org/P4310320595","host_organization_name":"Wiley","host_organization_lineage":["https://openalex.org/P4310320595"],"host_organization_lineage_names":["Wiley"],"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":"eJHaem","raw_type":"journal-article"},"sustainable_development_goals":[{"id":"https://metadata.un.org/sdg/3","score":0.98580002784729,"display_name":"Good health and well-being"}],"study_designs":[{"id":"https://openalex.org/study-designs/systematic-review","display_name":"Systematic Review"}],"awards":[],"funders":[{"id":"https://openalex.org/F4320307115","display_name":"Bristol-Myers Squibb","ror":"https://ror.org/00gtmwv55"},{"id":"https://openalex.org/F4320316629","display_name":"Agios Pharmaceuticals","ror":"https://ror.org/002x06r10"},{"id":"https://openalex.org/F4320336004","display_name":"Faculty of Medicine Siriraj Hospital, Mahidol University","ror":"https://ror.org/0331zs648"}],"has_content":{"grobid_xml":true,"pdf":true},"content_urls":{"pdf":"https://content.openalex.org/works/W4393900528.pdf","grobid_xml":"https://content.openalex.org/works/W4393900528.grobid-xml"},"referenced_works_count":25,"referenced_works":["https://openalex.org/W173608146","https://openalex.org/W1963786057","https://openalex.org/W1967354181","https://openalex.org/W1983552735","https://openalex.org/W2124925769","https://openalex.org/W2150644950","https://openalex.org/W2162821015","https://openalex.org/W2167575052","https://openalex.org/W2205989393","https://openalex.org/W2338611362","https://openalex.org/W2396599945","https://openalex.org/W2416285135","https://openalex.org/W2419658663","https://openalex.org/W2886674307","https://openalex.org/W2913121841","https://openalex.org/W2951790115","https://openalex.org/W3094483537","https://openalex.org/W3118615836","https://openalex.org/W3204966267","https://openalex.org/W4220996716","https://openalex.org/W4281614053","https://openalex.org/W4307965817","https://openalex.org/W4310118161","https://openalex.org/W4382011809","https://openalex.org/W4390545916"],"related_works":["https://openalex.org/W4310124579","https://openalex.org/W4283361304","https://openalex.org/W4365139473","https://openalex.org/W4385707454","https://openalex.org/W4283525236","https://openalex.org/W2983752881","https://openalex.org/W4389258268","https://openalex.org/W4382011809","https://openalex.org/W2906508065","https://openalex.org/W4365139596"],"abstract_inverted_index":{"A":[0],"recent":[1],"evidence":[2,62],"gaps":[3],"assessment":[4],"of":[5,10,33,71,77,100],"the":[6,28,34,44,54,68,81],"clinical,":[7],"health-related":[8],"quality":[9,99],"life,":[11],"and":[12,30,41,43,98],"economic":[13],"burden":[14,70,102],"associated":[15],"with":[16,64,93,109],"\u03b1-thalassemia":[17,72,94],"is":[18,104],"lacking.":[19],"We":[20],"conducted":[21],"a":[22,60],"systematic":[23],"literature":[24,52],"review":[25],"(SLR)":[26],"following":[27],"methodological":[29],"reporting":[31],"requirements":[32],"Preferred":[35],"Reporting":[36],"Items":[37],"for":[38,47],"Systematic":[39,48],"Reviews":[40],"Meta-Analyses":[42],"Cochrane":[45],"Handbook":[46],"Reviews,":[49],"using":[50],"available":[51,88],"over":[53],"past":[55,82],"decade.":[56],"This":[57],"SLR":[58],"identified":[59],"considerable":[61],"gap":[63],"regard":[65],"to":[66,107],"understanding":[67],"current":[69],"as":[73],"evident":[74],"from":[75],"paucity":[76],"studies":[78],"published":[79],"in":[80],"10":[83],"years.":[84],"The":[85],"limited":[86],"data":[87],"still":[89],"indicate":[90],"that":[91,103],"patients":[92,108],"experience":[95],"substantial":[96],"morbidity":[97],"life/economic":[101],"generally":[105],"comparable":[106],"\u03b2-thalassemia.":[110]},"counts_by_year":[{"year":2026,"cited_by_count":8},{"year":2025,"cited_by_count":3}],"updated_date":"2026-10-30T12:39:25.321706","created_date":"2024-04-04T00:00:00"}
