{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,19]],"date-time":"2026-03-19T11:36:46Z","timestamp":1773920206301,"version":"3.50.1"},"reference-count":80,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2026,3,19]],"date-time":"2026-03-19T00:00:00Z","timestamp":1773878400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Public Health"],"abstract":"<jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>Hemophilia is the most common severe hereditary bleeding disorder, with significant unmet needs in its management. This study aimed to assess the burden and unmet needs of severe hemophilia in Greece from a clinician and patient perspective.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Two independent Delphi panels were conducted: one comprising six clinical experts from all specialized hemophilia centers across Greece, and another including 15 patients-members of the Greek Hemophilia Society with severe hemophilia. Following a targeted literature review, two structured Delphi questionnaires were developed with evidence-based statements addressing the clinical, humanistic, and economic burden of severe hemophilia, as well as unmet medical needs, treatment preferences and expectations. Delphi panelists rated the statements over up to three rounds using a five-point Likert scale (from \u201cstrongly disagree\u201d to \u201cstrongly agree\u201d) to capture consensus and real-world perspectives. Consensus and its strength were established based on predefined criteria. Two focus group meetings, one with clinicians and one with patients, followed to discuss the Delphi findings in greater depth, validate areas of agreement or divergence, and collect qualitative insights.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Consensus was achieved on 97.3 and 97.2% of statements among clinicians and patients, respectively. The two groups showed largely concordant views, with limited areas of divergence. Both highlighted that, despite existing prophylactic treatments, spontaneous bleeds persist, significantly affecting the patients\u2019 and caregivers\u2019 quality of life and resulting in considerable non-medical and surgery costs. Suboptimal adherence remains a challenge, with main barriers including the frequency\/time commitment of factor replacement therapy, venous access challenges, and needle phobia. Both groups demonstrated the importance of safety in emerging treatments, highlighting their preference for treatments that support a more active lifestyle, while offering subcutaneous administration, superior efficacy, and greater convenience compared to the current standard of care.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Clinical and patient insights highlight the need for improved treatments for severe hemophilia in Greece. Safer, more effective therapies are needed to address residual burden and unmet needs. These should offer simpler administration, less frequent dosing, and more convenient delivery.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3389\/fpubh.2026.1766482","type":"journal-article","created":{"date-parts":[[2026,3,19]],"date-time":"2026-03-19T06:48:28Z","timestamp":1773902908000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Disease burden and unmet medical need in severe hemophilia in Greece: insights from clinicians and patients"],"prefix":"10.3389","volume":"14","author":[{"given":"Nikoletta","family":"Sofiaki","sequence":"first","affiliation":[{"name":"Pfizer Hellas","place":["Athens, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Oresteia","family":"Zisimopoulou","sequence":"additional","affiliation":[{"name":"Pfizer Hellas","place":["Athens, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Argyro","family":"Solakidi","sequence":"additional","affiliation":[{"name":"Pfizer Hellas","place":["Athens, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marina","family":"Economou","sequence":"additional","affiliation":[{"name":"1st Pediatric Department, Hippokration General Hospital, Aristotle University of Thessaloniki","place":["Thessaloniki, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Olga","family":"Katsarou","sequence":"additional","affiliation":[{"name":"Blood Unit and National Reference Centre for Congenital Bleeding Disorders, Laiko General Hospital","place":["Athens, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anna","family":"Kouramba","sequence":"additional","affiliation":[{"name":"Blood Unit and National Reference Centre for Congenital Bleeding Disorders, Laiko General Hospital","place":["Athens, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Efrosyni","family":"Nomikou","sequence":"additional","affiliation":[{"name":"Blood Bank and Hemophilia Unit, Hippokration General Hospital of Athens","place":["Athens, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Helen","family":"Pergantou","sequence":"additional","affiliation":[{"name":"Hemophilia Center\/Hemostasis and Thrombosis Unit, \u201cAgia Sofia\u201d Children\u2019s Hospital","place":["Athens, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sofia","family":"Vakalopoulou","sequence":"additional","affiliation":[{"name":"Hemophilia Center of Northern Greece, 2nd Propedeutic Department of Internal Medicine, Hippokration General Hospital of Thessaloniki, Aristotle University of Thessaloniki","place":["Thessaloniki, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"George","family":"Filippidis","sequence":"additional","affiliation":[{"name":"Greek Haemophilia Society","place":["Athens, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Vasilis","family":"Grammelis","sequence":"additional","affiliation":[{"name":"Pfizer Hellas","place":["Athens, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Damianos","family":"Menegas","sequence":"additional","affiliation":[{"name":"Pfizer Hellas","place":["Athens, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Andriani","family":"Angelopoulou","sequence":"additional","affiliation":[{"name":"Health Through Evidence","place":["Athens, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Charalampos","family":"Tzanetakos","sequence":"additional","affiliation":[{"name":"Health Through Evidence","place":["Athens, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"George","family":"Gourzoulidis","sequence":"additional","affiliation":[{"name":"Health Through Evidence","place":["Athens, Greece"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2026,3,19]]},"reference":[{"key":"ref1","volume-title":"Hemophilia","author":"Mehta","year":"2025"},{"key":"ref2","volume-title":"What is Hemophilia?","year":"2023"},{"key":"ref3","volume-title":"World Federation of Hemophilia Report on the Annual Global Survey 2022","year":"2023"},{"key":"ref4","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1111\/hae.14046","article-title":"WFH guidelines for the management of hemophilia","volume":"26","author":"Srivastava","year":"2020","journal-title":"Haemophilia"},{"key":"ref5","volume-title":"Hemophilia B","year":"2022"},{"key":"ref6","doi-asserted-by":"crossref","first-page":"459","DOI":"10.1586\/ehm.12.27","article-title":"Management of joint bleeding in hemophilia","volume":"5","author":"Simpson","year":"2012","journal-title":"Expert Rev Hematol"},{"key":"ref7","doi-asserted-by":"crossref","first-page":"51","DOI":"10.15256\/joc.2011.1.2","article-title":"Haemophilia and joint disease: pathophysiology, evaluation, and management","volume":"1","author":"Knobe","year":"2011","journal-title":"J Comorb"},{"key":"ref8","doi-asserted-by":"crossref","first-page":"e181","DOI":"10.1055\/s-0044-1785524","article-title":"Disease burden, clinical outcomes, and quality of life in people with Hemophilia a without inhibitors in Europe: analyses from CHESS II\/CHESS PAEDs","volume":"8","author":"Chowdary","year":"2024","journal-title":"TH Open"},{"key":"ref9","doi-asserted-by":"crossref","first-page":"557","DOI":"10.1111\/hae.14571","article-title":"Pain, depression and anxiety in people with haemophilia from three Nordic countries: cross-sectional survey data from the MIND study","volume":"28","author":"Steen Carlsson","year":"2022","journal-title":"Haemophilia"},{"key":"ref10","volume-title":"Treatment of Hemophilia","year":"2023"},{"key":"ref11","doi-asserted-by":"crossref","first-page":"100890","DOI":"10.1016\/j.blre.2021.100890","article-title":"Hemophilia treatment in 2021: choosing the \"optimal\" treatment using an integrative, patient-oriented approach to shared decision-making between patients and clinicians","volume":"52","author":"Hermans","year":"2022","journal-title":"Blood Rev"},{"key":"ref12","year":"2024"},{"key":"ref13","doi-asserted-by":"crossref","first-page":"75","DOI":"10.1007\/s40261-021-01108-4","article-title":"Application of multicriteria decision analysis to determine the value of prophylaxis relative to on-demand treatment in Hemophilia a and Emicizumab versus replacement therapy in the Greek healthcare setting","volume":"42","author":"Gourzoulidis","year":"2022","journal-title":"Clin Drug Investig"},{"key":"ref14","doi-asserted-by":"crossref","first-page":"237","DOI":"10.1586\/17474086.2015.1002465","article-title":"Individualizing prophylaxis in hemophilia: a review","volume":"8","author":"Petrini","year":"2015","journal-title":"Expert Rev Hematol"},{"key":"ref15","doi-asserted-by":"crossref","first-page":"339","DOI":"10.1111\/ejh.14135","article-title":"The current challenges faced by people with hemophilia B","volume":"112","author":"Miesbach","year":"2024","journal-title":"Eur J Haematol"},{"key":"ref16","doi-asserted-by":"crossref","first-page":"629","DOI":"10.2147\/JBM.S446204","article-title":"Current status and challenges in delivering Comprehensive Care for Patients with Hemophilia","volume":"14","author":"Nomura","year":"2023","journal-title":"J Blood Med"},{"key":"ref17","doi-asserted-by":"crossref","first-page":"2314","DOI":"10.1182\/blood-2024-204490","article-title":"Unmet needs in people with Hemophilia receiving prophylaxis treatment: a real-world survey","volume":"144","author":"Malec","year":"2024","journal-title":"Blood"},{"key":"ref18","doi-asserted-by":"crossref","first-page":"e61","DOI":"10.59556\/japi.73.1055","article-title":"Unmet needs in Hemophilia: a narrative review","volume":"73","author":"Jena","year":"2025","journal-title":"J Assoc Physicians India"},{"key":"ref19","doi-asserted-by":"crossref","first-page":"23","DOI":"10.1016\/j.jclinepi.2019.03.011","article-title":"Delphi procedure in core outcome set development: rating scale and consensus criteria determined outcome selection","volume":"111","author":"De Meyer","year":"2019","journal-title":"J Clin Epidemiol"},{"key":"ref20","doi-asserted-by":"crossref","first-page":"254","DOI":"10.1111\/hae.14504","article-title":"Haemophilia management and treatment: an Italian survey on patients', caregivers' and clinicians' point of view","volume":"28","author":"Cortesi","year":"2022","journal-title":"Haemophilia"},{"key":"ref21","doi-asserted-by":"crossref","first-page":"954","DOI":"10.1111\/hae.14803","article-title":"Bleeding events in people with congenital haemophilia a without factor VIII inhibitors receiving prophylactic factor VIII treatment: a systematic literature review","volume":"29","author":"Mannucci","year":"2023","journal-title":"Haemophilia"},{"key":"ref22","doi-asserted-by":"crossref","first-page":"697","DOI":"10.3390\/life14060697","article-title":"A retrospective observational study of quality of life in a Northern Greece population of people with haemophilia","volume":"14","author":"Moka","year":"2024","journal-title":"Life"},{"key":"ref23","doi-asserted-by":"crossref","first-page":"815","DOI":"10.1177\/1076029617733041","article-title":"Health-related quality of life and association with arthropathy in Greek patients with Hemophilia","volume":"24","author":"Varaklioti","year":"2018","journal-title":"Clin Appl Thromb Hemost"},{"key":"ref24","doi-asserted-by":"crossref","first-page":"433","DOI":"10.1111\/hae.13754","article-title":"From the voices of people with haemophilia a and their caregivers: challenges with current treatment, their impact on quality of life and desired improvements in future therapies","volume":"25","author":"Wiley","year":"2019","journal-title":"Haemophilia"},{"key":"ref25","doi-asserted-by":"crossref","first-page":"3300","DOI":"10.3390\/jcm14103300","article-title":"Patient-centred management of well-controlled haemophilia: obtaining opinions and definitions through a Delphi consensus","volume":"14","author":"Berrueco","year":"2025","journal-title":"J Clin Med"},{"key":"ref26","first-page":"647","article-title":"Health care costs and resource use of managing hemophilia a: a targeted literature review","volume":"29","author":"Chen","year":"2023","journal-title":"J Manag Care Spec Pharm"},{"key":"ref27","doi-asserted-by":"crossref","first-page":"938","DOI":"10.1111\/hae.14302","article-title":"Clinical attributes and treatment characteristics are associated with work productivity and activity impairment in people with severe haemophilia a","volume":"27","author":"O'Hara","year":"2021","journal-title":"Haemophilia"},{"key":"ref28","doi-asserted-by":"crossref","first-page":"398","DOI":"10.1111\/hae.14270","article-title":"Health-related quality of life and health status in adolescent and adult people with haemophilia a without factor VIII inhibitors-a non-interventional study","volume":"27","author":"Oldenburg","year":"2021","journal-title":"Haemophilia"},{"key":"ref29","doi-asserted-by":"crossref","first-page":"e2020001","DOI":"10.4084\/mjhid.2020.001","article-title":"Molecular mechanisms of inhibitor development in Hemophilia","volume":"12","author":"Tieu","year":"2020","journal-title":"Mediterr J Hematol Infect Diseases"},{"key":"ref30","doi-asserted-by":"crossref","first-page":"904","DOI":"10.1055\/s-0042-1756188","article-title":"The more recent history of Hemophilia treatment","volume":"48","author":"Franchini","year":"2022","journal-title":"Semin Thromb Hemost"},{"key":"ref31","first-page":"221","article-title":"Recent advances in the treatment of Hemophilia: a review","volume":"15","author":"Marchesini","year":"2021","journal-title":"Biol: Targets Therapy"},{"key":"ref32","doi-asserted-by":"crossref","first-page":"S634","DOI":"10.1016\/j.jval.2024.10.3181","article-title":"SA100 effect of adherence to Hemophilia drug therapy on outcomes: a systematic literature review","volume":"27","author":"Yubin Sohn","year":"2024","journal-title":"Value Health"},{"key":"ref33","doi-asserted-by":"crossref","first-page":"413","DOI":"10.1111\/j.1538-7836.2008.03270.x","article-title":"Break-through bleeding in relation to predicted factor VIII levels in patients receiving prophylactic treatment for severe hemophilia a","volume":"7","author":"Collins","year":"2009","journal-title":"J Thromb Haemost"},{"key":"ref34","doi-asserted-by":"crossref","first-page":"458","DOI":"10.1111\/hae.12618","article-title":"Adherence to prophylaxis and quality of life in children and adolescents with severe haemophilia a","volume":"21","author":"Garcia-Dasi","year":"2015","journal-title":"Haemophilia"},{"key":"ref35","first-page":"152","article-title":"Physical activity improved by adherence to prophylaxis in an Italian population of children, adolescents and adults with severe haemophilia a: the SHAPE study","volume":"18","author":"Zanon","year":"2020","journal-title":"Blood Transfus"},{"key":"ref36","doi-asserted-by":"crossref","first-page":"64","DOI":"10.1111\/hae.12533","article-title":"Adherence to prophylaxis is associated with better outcomes in moderate and severe haemophilia: results of a patient survey","volume":"21","author":"Krishnan","year":"2015","journal-title":"Haemophilia"},{"key":"ref37","doi-asserted-by":"crossref","first-page":"71","DOI":"10.1007\/s40267-022-00967-x","article-title":"Treatment satisfaction and limitations in haemophilia a, with a focus on factor VIII product storage conditions: patients\u2019 perspectives and challenges","volume":"39","author":"Zupan","year":"2022","journal-title":"Drugs Ther Perspect"},{"key":"ref38","doi-asserted-by":"crossref","first-page":"1702","DOI":"10.3390\/healthcare9121702","article-title":"Medication adherence for haemophilia patients: outcome of prophylaxis treatment intervention","volume":"9","author":"Mokhtar","year":"2021","journal-title":"Healthcare (Basel)"},{"key":"ref39","doi-asserted-by":"crossref","first-page":"318","DOI":"10.1002\/rth2.12301","article-title":"Hemophilia prophylaxis adherence and bleeding using a tailored, frequency-escalated approach: the Canadian Hemophilia primary prophylaxis study","volume":"4","author":"Dover","year":"2020","journal-title":"Res Pract Thromb Haemost"},{"key":"ref40","doi-asserted-by":"crossref","first-page":"337","DOI":"10.1080\/13696998.2016.1265973","article-title":"Continuous prophylaxis with recombinant factor IX fc fusion protein and conventional recombinant factor IX products: comparisons of efficacy and weekly factor consumption","volume":"20","author":"Iorio","year":"2017","journal-title":"J Med Econ"},{"key":"ref41","doi-asserted-by":"crossref","first-page":"S3","DOI":"10.1002\/ajh.24220","article-title":"Treatment outcomes, quality of life, and impact of hemophilia on young adults (aged 18-30 years) with hemophilia","volume":"90","author":"Witkop","year":"2015","journal-title":"Am J Hematol"},{"key":"ref42","doi-asserted-by":"crossref","first-page":"821","DOI":"10.1111\/hae.13811","article-title":"Comparing objective and self-reported measures of adherence in haemophilia","volume":"25","author":"Guedes","year":"2019","journal-title":"Haemophilia"},{"key":"ref43","doi-asserted-by":"crossref","first-page":"148","DOI":"10.1179\/1607845414Y.0000000176","article-title":"Adherence to clotting factors among persons with hemophilia a or B","volume":"20","author":"Armstrong","year":"2015","journal-title":"Hematology"},{"key":"ref44","doi-asserted-by":"crossref","first-page":"566","DOI":"10.1111\/hae.13221","article-title":"Haemophilia patients' unmet needs and their expectations of the new extended half-life factor concentrates","volume":"23","author":"von Mackensen","year":"2017","journal-title":"Haemophilia"},{"key":"ref45","doi-asserted-by":"crossref","first-page":"431","DOI":"10.2147\/PPA.S151812","article-title":"Patient preferences in the treatment of hemophilia a: impact of storage conditions on product choice","volume":"12","author":"Tischer","year":"2018","journal-title":"Patient Prefer Adherence"},{"key":"ref46","doi-asserted-by":"crossref","first-page":"1677","DOI":"10.2147\/PPA.S139851","article-title":"Treatment adherence in hemophilia","volume":"11","author":"Thornburg","year":"2017","journal-title":"Patient Prefer Adherence"},{"key":"ref47","doi-asserted-by":"crossref","first-page":"1640","DOI":"10.1016\/j.jtha.2024.02.009","article-title":"Comparison of thrombotic adverse events in patients treated with factor VIII products and emicizumab using the 2018-2022 US food and drug administration adverse event reporting system data","volume":"22","author":"Cho","year":"2024","journal-title":"J Thromb Haemost"},{"key":"ref48","year":"2018"},{"key":"ref49","first-page":"673","article-title":"Challenges associated with access to recently developed hemophilia treatments in routine care: perspectives of healthcare professionals","volume":"110","author":"Berger","year":"2025","journal-title":"Haematologica"},{"key":"ref50","doi-asserted-by":"crossref","DOI":"10.3389\/fmed.2025.1618464","article-title":"Therapeutic advances in hemophilia: from molecular innovation to patient-centered global care","volume":"12","author":"Dushimova","year":"2025","journal-title":"Front Med"},{"key":"ref51","doi-asserted-by":"crossref","first-page":"1623","DOI":"10.2147\/PPA.S92985","article-title":"Understanding patient preferences and willingness to pay for hemophilia therapies","volume":"9","author":"Chaugule","year":"2015","journal-title":"Patient Prefer Adherence"},{"key":"ref52","doi-asserted-by":"crossref","first-page":"266","DOI":"10.1186\/s12876-025-03867-2","article-title":"Cost-effectiveness of NALIRIFOX versus nab-paclitaxel and gemcitabine in previously untreated metastatic pancreatic ductal adenocarcinoma","volume":"25","author":"Liu","year":"2025","journal-title":"BMC Gastroenterol"},{"key":"ref53","doi-asserted-by":"crossref","first-page":"1121","DOI":"10.2217\/cer-2021-0109","article-title":"Real-world treatment, clinical outcomes and healthcare resource utilization among persons with hemophilia a by age","volume":"10","author":"Caplan","year":"2021","journal-title":"J Comp Eff Res"},{"key":"ref54","doi-asserted-by":"crossref","first-page":"143","DOI":"10.1186\/s13023-021-01774-9","article-title":"Clinical, humanistic, and economic burden of severe hemophilia B in the United States: results from the CHESS US and CHESS US+ population surveys","volume":"16","author":"Burke","year":"2021","journal-title":"Orphanet J Rare Dis"},{"key":"ref55","doi-asserted-by":"crossref","first-page":"271","DOI":"10.1111\/hae.13398","article-title":"Prevalence of and risk factors for cerebral microbleeds among adult patients with haemophilia a or B","volume":"24","author":"Husseinzadeh","year":"2018","journal-title":"Haemophilia"},{"key":"ref56","doi-asserted-by":"crossref","DOI":"10.3390\/jcm12041368","article-title":"Mild and moderate hemophilia a: neglected conditions, still with unmet needs","volume":"12","author":"Castaman","year":"2023","journal-title":"J Clin Med"},{"key":"ref57","doi-asserted-by":"crossref","first-page":"165","DOI":"10.1302\/2058-5241.4.180090","article-title":"The role of orthopaedic surgery in haemophilia: current rationale, indications and results","volume":"4","author":"Rodr\u00edguez-Merch\u00e1n","year":"2019","journal-title":"EFORT Open Rev"},{"key":"ref58","doi-asserted-by":"crossref","first-page":"995","DOI":"10.3892\/etm.2021.10427","article-title":"Orthopedic surgery in hemophilic patients with musculoskeletal disorders: a systematic review","volume":"22","author":"Badulescu","year":"2021","journal-title":"Exp Ther Med"},{"key":"ref59","doi-asserted-by":"crossref","first-page":"1969","DOI":"10.3390\/jcm11071969","article-title":"Intracranial haemorrhage in haemophilia patients is still an open issue: the final results of the Italian EMO.REC Registry","volume":"11","author":"Zanon","year":"2022","journal-title":"J Clin Med"},{"key":"ref60","doi-asserted-by":"crossref","first-page":"532","DOI":"10.1111\/hae.14557","article-title":"Associated comorbidities, healthcare utilization & mortality in hospitalized patients with haemophilia in the United States: contemporary nationally representative estimates","volume":"28","author":"Day","year":"2022","journal-title":"Haemophilia"},{"key":"ref61","doi-asserted-by":"crossref","first-page":"645","DOI":"10.1111\/jth.15182","article-title":"Mortality, life expectancy, and causes of death of persons with hemophilia in the Netherlands 2001-2018","volume":"19","author":"Hassan","year":"2021","journal-title":"J Thromb Haemost"},{"key":"ref62","doi-asserted-by":"crossref","first-page":"111","DOI":"10.1111\/ejh.13193","article-title":"Inhibitors in haemophilia a and B: management of bleeds, inhibitor eradication and strategies for difficult-to-treat patients","volume":"102","author":"Ljung","year":"2019","journal-title":"Eur J Haematol"},{"key":"ref63","doi-asserted-by":"crossref","first-page":"e322","DOI":"10.1111\/hae.13596","article-title":"Real-world comparative analysis of bleeding complications and health-related quality of life in patients with haemophilia a and haemophilia B","volume":"24","author":"Booth","year":"2018","journal-title":"Haemophilia"},{"key":"ref64","doi-asserted-by":"crossref","first-page":"1042","DOI":"10.1007\/s00508-020-01798-4","article-title":"Pain management in hemophilia: expert recommendations","volume":"133","author":"Stromer","year":"2021","journal-title":"Wien Klin Wochenschr"},{"key":"ref65","doi-asserted-by":"crossref","first-page":"1577","DOI":"10.2147\/PPA.S212723","article-title":"Interrelationship between depression, anxiety, pain, and treatment adherence in hemophilia: results from a US cross-sectional survey","volume":"13","author":"Witkop","year":"2019","journal-title":"Patient Prefer Adherence"},{"key":"ref66","doi-asserted-by":"crossref","first-page":"521","DOI":"10.1186\/s13023-021-02152-1","article-title":"Clinical, humanistic, and economic burden of severe haemophilia B in adults receiving factor IX prophylaxis: findings from the CHESS II real-world burden of illness study in Europe","volume":"16","author":"Burke","year":"2021","journal-title":"Orphanet J Rare Dis"},{"key":"ref67","doi-asserted-by":"crossref","first-page":"424","DOI":"10.1111\/hae.13684","article-title":"The impact of psychosocial determinants on caregivers' burden of children with haemophilia (results of the BBC study)","volume":"25","author":"von Mackensen","year":"2019","journal-title":"Haemophilia"},{"key":"ref68","first-page":"1095","article-title":"Assessing health care resource use, outcomes, and costs among Medicaid beneficiaries receiving factor IX prophylaxis for hemophilia B","volume":"30","author":"Pauly","year":"2024","journal-title":"J Manag Care Spec Pharm"},{"key":"ref69","doi-asserted-by":"crossref","first-page":"341","DOI":"10.1002\/jha2.412","article-title":"Resource utilization and treatment costs of patients with severe hemophilia a: real-world data from the ATHNdataset","volume":"3","author":"Recht","year":"2022","journal-title":"EJHaem"},{"key":"ref70","doi-asserted-by":"crossref","first-page":"e157","DOI":"10.37765\/ajmc.2024.89543","article-title":"Bleeds and resource use in hemophilia B: retrospective observational study","volume":"30","author":"Young","year":"2024","journal-title":"Am J Manag Care"},{"key":"ref71","doi-asserted-by":"crossref","first-page":"457","DOI":"10.3111\/13696998.2015.1016228","article-title":"Burden of illness: direct and indirect costs among persons with hemophilia a in the United States","volume":"18","author":"Zhou","year":"2015","journal-title":"J Med Econ"},{"key":"ref72","doi-asserted-by":"crossref","first-page":"300060519862101","DOI":"10.1177\/0300060519862101","article-title":"Challenges facing community-dwelling adults with hemophilia: implications for community-based adult education and nursing","volume":"48","author":"Okide","year":"2020","journal-title":"J Int Med Res"},{"key":"ref73","doi-asserted-by":"crossref","DOI":"10.3390\/jcm13175121","article-title":"Teenagers and adolescents with Hemophilia-need for a specific approach","volume":"13","author":"K\u00f6nigs","year":"2024","journal-title":"J Clin Med"},{"key":"ref74","doi-asserted-by":"crossref","first-page":"187","DOI":"10.1111\/hae.15153","article-title":"Systematic literature review of outcomes associated with adherence to haemophilia drug therapy","volume":"31","author":"Khanji","year":"2025","journal-title":"Haemophilia"},{"key":"ref75","article-title":"The dosing conundrum of emicizumab: to waste product or not? Research and practice","volume":"7","author":"Young","year":"2023","journal-title":"Thromb Haemost"},{"key":"ref76","doi-asserted-by":"crossref","first-page":"55","DOI":"10.1080\/21642850.2020.1718501","article-title":"Adherence to prophylaxis in adolescents and young adults with severe haemophilia: a qualitative study with healthcare professionals","volume":"8","author":"van Os","year":"2020","journal-title":"Health Psychol Behav Med"},{"key":"ref77","doi-asserted-by":"crossref","first-page":"375","DOI":"10.1111\/hae.14928","article-title":"Patient and caregiver preferences for haemophilia treatments: a discrete-choice experiment","volume":"30","author":"Garcia","year":"2024","journal-title":"Haemophilia"},{"key":"ref78","doi-asserted-by":"crossref","first-page":"2591","DOI":"10.1182\/blood-2024-207425","article-title":"Patient and caregiver preferences for Hemophilia prophylactic treatments: a discrete choice experiment","volume":"144","author":"Lu","year":"2024","journal-title":"Blood"},{"key":"ref79","doi-asserted-by":"crossref","first-page":"100890","DOI":"10.1016\/j.blre.2021.100890","article-title":"Hemophilia treatment in 2021: choosing the\"optimal\" treatment using an integrative, patient-oriented approach to shared decision-making between patients and clinicians","volume":"52","author":"Hermans","year":"2022","journal-title":"Blood Rev"},{"key":"ref80","doi-asserted-by":"crossref","first-page":"101035","DOI":"10.1016\/j.vhri.2024.101035","article-title":"Patient and caregiver preferences for Hemophilia a treatment in Taiwan: a discrete choice experiment","volume":"45","author":"Chiou","year":"2025","journal-title":"Value Health Reg Issues"}],"container-title":["Frontiers in Public Health"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fpubh.2026.1766482\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,3,19]],"date-time":"2026-03-19T06:48:36Z","timestamp":1773902916000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fpubh.2026.1766482\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,3,19]]},"references-count":80,"alternative-id":["10.3389\/fpubh.2026.1766482"],"URL":"https:\/\/doi.org\/10.3389\/fpubh.2026.1766482","relation":{},"ISSN":["2296-2565"],"issn-type":[{"value":"2296-2565","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,3,19]]},"article-number":"1766482"}}