{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,10]],"date-time":"2026-04-10T22:10:29Z","timestamp":1775859029932,"version":"3.50.1"},"reference-count":60,"publisher":"Public Library of Science (PLoS)","issue":"4","license":[{"start":{"date-parts":[[2022,4,28]],"date-time":"2022-04-28T00:00:00Z","timestamp":1651104000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000050","name":"National Heart, Lung, and Blood Institute","doi-asserted-by":"publisher","award":["OT3HL152448"],"award-info":[{"award-number":["OT3HL152448"]}],"id":[{"id":"10.13039\/100000050","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000050","name":"National Heart, Lung, and Blood Institute","doi-asserted-by":"publisher","award":["OT3HL151434"],"award-info":[{"award-number":["OT3HL151434"]}],"id":[{"id":"10.13039\/100000050","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS ONE"],"abstract":"<jats:sec id=\"sec001\">\n<jats:title>Background<\/jats:title>\n<jats:p>Sickle cell disease (SCD) is a clinically heterogeneous disease with many acute and chronic complications driven by ongoing vaso-occlusion and hemolysis. It causes a disproportionate burden on Black and Hispanic communities. Our objective was to follow the SMDM\/ISPOR Task Force recommendations for good practices and create a conceptual model of the progression of SCD under current clinical practice to inform cost-effectiveness analyses (CEA) of promising curative therapies in the pipeline over a lifetime horizon.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"sec002\">\n<jats:title>Methods<\/jats:title>\n<jats:p>We used consultations with experts, providers, and patients to identify acute events and chronic conditions in the conceptual model. We compared our model structure to previous CEA models of interventions for SCD, assessed the prevalence of the identified disease attributes in Medicaid and Medicare claims databases, and identified relevant outcomes following the 2<jats:sup>nd<\/jats:sup> Panel in CEA. We determined an appropriate modeling technique and relevant data sources for parameterizing the model.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"sec003\">\n<jats:title>Results<\/jats:title>\n<jats:p>The conceptual model structure included four dimensions of disease: chronic pain, acute events, chronic conditions, and treatment complications, spanning 26 disease attributes with significant impacts on health-related quality of life and resource. We modeled chronic pain separately to reflect its importance to patients and interaction with all other disease attributes. We identified additional data sources for health state utilities and non-medical costs and benefits of SCD. We will use a microsimulation model with age- and sex-specific transitions between health states predicted by patient demographic characteristics and disease history.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"sec004\">\n<jats:title>Conclusion<\/jats:title>\n<jats:p>Developing the model structure through an explicit process of model conceptualization can increase the transparency and accuracy of results. We will populate the conceptual model with the data sources described and evaluate the cost-effectiveness of curative therapies.<\/jats:p>\n<\/jats:sec>","DOI":"10.1371\/journal.pone.0267448","type":"journal-article","created":{"date-parts":[[2022,4,28]],"date-time":"2022-04-28T19:00:36Z","timestamp":1651172436000},"page":"e0267448","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":6,"title":["Development of a conceptual model for evaluating new non-curative and curative therapies for sickle cell disease"],"prefix":"10.1371","volume":"17","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7406-2448","authenticated-orcid":true,"given":"Kate M.","family":"Johnson","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Boshen","family":"Jiao","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"M. A.","family":"Bender","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Scott D.","family":"Ramsey","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Beth","family":"Devine","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4238-7402","authenticated-orcid":true,"given":"Anirban","family":"Basu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"340","published-online":{"date-parts":[[2022,4,28]]},"reference":[{"key":"pone.0267448.ref001","author":"Centers for Disease Control and Prevention","year":"2020","journal-title":"Data & Statistics on Sickle Cell Disease"},{"key":"pone.0267448.ref002","doi-asserted-by":"crossref","first-page":"311","DOI":"10.1016\/S0140-6736(17)30193-9","article-title":"Sickle cell disease","volume":"390","author":"RE Ware","year":"2017","journal-title":"The Lancet"},{"key":"pone.0267448.ref003","doi-asserted-by":"crossref","first-page":"377","DOI":"10.1002\/pbc.24176","article-title":"Health-related quality of life in sickle cell disease: Past, present, and future.","volume":"59","author":"JA Panepinto","year":"2012","journal-title":"Pediatric Blood & Cancer."},{"key":"pone.0267448.ref004","doi-asserted-by":"crossref","first-page":"176","DOI":"10.1186\/s12955-018-1009-5","article-title":"Quality of life among caregivers of sickle cell disease patients: a cross sectional study","volume":"16","author":"BM Madani","year":"2018","journal-title":"Health and Quality of Life Outcomes"},{"key":"pone.0267448.ref005","doi-asserted-by":"crossref","DOI":"10.1001\/jamanetworkopen.2019.15374","article-title":"Estimated Life Expectancy and Income of Patients With Sickle Cell Disease Compared With Those Without Sickle Cell Disease.","volume":"2","author":"D Lubeck","year":"2019","journal-title":"JAMA Netw Open"},{"key":"pone.0267448.ref006","doi-asserted-by":"crossref","first-page":"1095","DOI":"10.1016\/j.jval.2021.02.014","article-title":"Indirect Economic Burden of Sickle Cell Disease.","volume":"24","author":"D Holdford","year":"2021","journal-title":"Value in Health"},{"key":"pone.0267448.ref007","doi-asserted-by":"crossref","first-page":"621","DOI":"10.1016\/j.jval.2019.03.014","article-title":"Estimating the Clinical Pipeline of Cell and Gene Therapies and Their Potential Economic Impact on the US Healthcare System.","volume":"22","author":"C Quinn","year":"2019","journal-title":"Value in Health"},{"key":"pone.0267448.ref008","doi-asserted-by":"crossref","first-page":"515","DOI":"10.1111\/bjh.14167","article-title":"Haematopoietic stem cell transplantation for sickle cell disease\u2013current practice and new approaches","volume":"174","author":"SD Arnold","year":"2016","journal-title":"British Journal of Haematology"},{"key":"pone.0267448.ref009","doi-asserted-by":"crossref","first-page":"1810","DOI":"10.1016\/j.mayocp.2018.08.001","article-title":"Advances in the Treatment of Sickle Cell Disease","volume":"93","author":"S Kapoor","year":"2018","journal-title":"Mayo Clinic Proceedings"},{"key":"pone.0267448.ref010","doi-asserted-by":"crossref","first-page":"1102","DOI":"10.1038\/d41587-019-00026-3","article-title":"First gene therapy for \u03b2-thalassemia approved","volume":"37","author":"C. 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Varni","journal-title":"The PedsQL TM\u2014Measurement Model for the Pediatric Quality of Life inventory"},{"key":"pone.0267448.ref037","article-title":"Mapping and validation of generic PedsQL scores to utility values for individuals with sickle cell disease","author":"B Jiao","year":"2021","journal-title":"Quality of Life Research"},{"key":"pone.0267448.ref038","article-title":"Health State Utilities for Sickle Cell Disease: A Catalog Prepared From a Systematic Review.","author":"B Jiao","year":"2021","journal-title":"Value in Health"},{"key":"pone.0267448.ref039","volume-title":"Estimating Costs and Valuations of Non-Health Benefits in Cost-Effectiveness Analysis.","author":"A Basu","year":"2016","edition":"2"},{"key":"pone.0267448.ref040","volume-title":"Associating Health-Related Quality of Life with Time Use in the United States to Inform Productivity and Time Costs Calculation in Cost-Effectiveness Analysis","author":"Bohsen Jiao"},{"key":"pone.0267448.ref041","doi-asserted-by":"crossref","first-page":"957","DOI":"10.1016\/j.jval.2021.03.006","article-title":"Catalog of Age- and Medical Condition\u2014Specific Healthcare Costs in the United States to Inform Future Costs Calculations in Cost-Effectiveness Analysis.","volume":"24","author":"B Jiao","year":"2021","journal-title":"Value in Health"},{"key":"pone.0267448.ref042","article-title":"Medical and Non-Medical Costs of Sickle Cell Disease and Treatments: A Systematic Review and Landscape Analysis.","author":"Zach Baldwin","journal-title":"PharmacoEconomics\u2013Open"},{"key":"pone.0267448.ref043","doi-asserted-by":"crossref","first-page":"879","DOI":"10.1182\/blood.V85.4.879.bloodjournal854879","article-title":"Neurologic complications after allogeneic marrow transplantation for sickle cell anemia [see comments]","volume":"85","author":"M Walters","year":"1995","journal-title":"Blood"},{"key":"pone.0267448.ref044","doi-asserted-by":"crossref","first-page":"e620","DOI":"10.3399\/bjgp11X601325","article-title":"Screening for sickle cell and thalassaemia in primary care: a cost-effectiveness study","volume":"61","author":"S Bryan","year":"2011","journal-title":"Br J Gen Pract"},{"key":"pone.0267448.ref045","doi-asserted-by":"crossref","first-page":"2034","DOI":"10.1111\/trf.12585","article-title":"Economic Evaluation of a Hypothetical Screening Assay for Alloimmunization Risk Among Transfused Patients with Sickle Cell Disease","volume":"54","author":"S Kacker","year":"2014","journal-title":"Transfusion"},{"key":"pone.0267448.ref046","doi-asserted-by":"crossref","first-page":"304","DOI":"10.1186\/s12913-016-1572-6","article-title":"Newborn screening and prophylactic interventions for sickle cell disease in 47 countries in sub-Saharan Africa: a cost-effectiveness analysis.","volume":"16","author":"A Kuznik","year":"2016","journal-title":"BMC Health Serv Res."},{"key":"pone.0267448.ref047","doi-asserted-by":"crossref","first-page":"1","DOI":"10.3310\/hta13010","article-title":"Deferasirox for the treatment of iron overload associated with regular blood transfusions (transfusional hemosiderosis) in patients suffering with chronic anaemia: a systematic review and economic evaluation.","volume":"13","author":"C McLeod","year":"2009","journal-title":"Health Technology Assessment."},{"key":"pone.0267448.ref048","volume-title":"The clinical effectiveness and cost-effectiveness of primary stroke prevention in children with sickle cell disease: a systematic review and economic evaluation. NIHR Health Technology Assessment programme: Executive Summaries","author":"MG Cherry","year":"2012"},{"key":"pone.0267448.ref049","article-title":"Crizanlizumab, Voxelotor, and L-Glutamine for Sickle Cell Disease: Effectiveness and Value.","author":"P Bradt","year":"2020","journal-title":"Institute for Clinical and Economic Review"},{"key":"pone.0267448.ref050","doi-asserted-by":"crossref","first-page":"1314","DOI":"10.1016\/j.jpeds.2015.08.068","article-title":"A Cost-Effectiveness Analysis of a Pilot Neonatal Screening Program for Sickle Cell Anemia in the Republic of Angola","volume":"167","author":"PT McGann","year":"2015","journal-title":"J Pediatr"},{"key":"pone.0267448.ref051","doi-asserted-by":"crossref","first-page":"10838","DOI":"10.1038\/s41598-021-90405-1","article-title":"Cost-effectiveness of a hypothetical cell or gene therapy cure for sickle cell disease","volume":"11","author":"J Salcedo","year":"2021","journal-title":"Sci Rep"},{"key":"pone.0267448.ref052","first-page":"183","article-title":"Understanding pain and improving management of sickle cell disease: the PiSCES study","volume":"97","author":"WR Smith","year":"2005","journal-title":"J Natl Med Assoc"},{"key":"pone.0267448.ref053","doi-asserted-by":"crossref","first-page":"562","DOI":"10.1182\/hematology.2020000143","article-title":"Optimizing the management of chronic pain in sickle cell disease","volume":"2020","author":"I Osunkwo","year":"2020","journal-title":"Hematology"},{"key":"pone.0267448.ref054","doi-asserted-by":"crossref","first-page":"94","DOI":"10.7326\/0003-4819-148-2-200801150-00004","article-title":"Daily assessment of pain in adults with sickle cell disease","volume":"148","author":"WR Smith","year":"2008","journal-title":"Ann Intern Med"},{"key":"pone.0267448.ref055","doi-asserted-by":"crossref","first-page":"1033","DOI":"10.1001\/jama.2014.10517","article-title":"Management of Sickle Cell Disease: Summary of the 2014 Evidence-Based Report by Expert Panel Members","volume":"312","author":"BP Yawn","year":"2014","journal-title":"JAMA"},{"key":"pone.0267448.ref056","volume-title":"Long-term Follow-up after Hematopoietic Stem Cell Transplant General Guidelines for Referring Physicians.","year":"2021"},{"key":"pone.0267448.ref057","doi-asserted-by":"crossref","first-page":"1345","DOI":"10.1080\/13696998.2020.1813144","article-title":"Vaso-occlusive crises and costs of sickle cell disease in patients with commercial, Medicaid, and Medicare insurance\u2013the perspective of private and public payers.","volume":"23","author":"NR Shah","year":"2020","journal-title":"Journal of Medical Economics."},{"key":"pone.0267448.ref058","article-title":"Landscape Analysis of Gene-based Trials for the Treatment of Sickle Cell Disease: Preparing to Estimate Value.","author":"D Quach","year":"2021","journal-title":"Expert Review of Pharmacoeconomics and Outcomes Research"},{"key":"pone.0267448.ref059","article-title":"Bluebird prices gene therapy at 1.58 million euros over 5 years.","author":"D Beasley","year":"2019","journal-title":"Reuters Health News."},{"key":"pone.0267448.ref060","doi-asserted-by":"crossref","first-page":"727","DOI":"10.1007\/s40273-017-0509-1","article-title":"Emerging Use of Early Health Technology Assessment in Medical Product Development: A Scoping Review of the Literature.","volume":"35","author":"MJ IJzerman","year":"2017","journal-title":"Pharmacoeconomics"}],"container-title":["PLOS ONE"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/dx.plos.org\/10.1371\/journal.pone.0267448","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,4,28]],"date-time":"2022-04-28T19:01:54Z","timestamp":1651172514000},"score":1,"resource":{"primary":{"URL":"https:\/\/dx.plos.org\/10.1371\/journal.pone.0267448"}},"subtitle":[],"editor":[{"given":"Mohamed A.","family":"Yassin","sequence":"first","affiliation":[],"role":[{"role":"editor","vocabulary":"crossref"}]}],"short-title":[],"issued":{"date-parts":[[2022,4,28]]},"references-count":60,"journal-issue":{"issue":"4","published-online":{"date-parts":[[2022,4,28]]}},"URL":"https:\/\/doi.org\/10.1371\/journal.pone.0267448","relation":{},"ISSN":["1932-6203"],"issn-type":[{"value":"1932-6203","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,4,28]]}}}