{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T05:49:14Z","timestamp":1787032154010,"version":"build-2736575974"},"reference-count":19,"publisher":"Becaris Publishing Limited","issue":"4","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J. Comp. Eff. Res."],"published-print":{"date-parts":[[2025,4]]},"abstract":"<jats:p>\n                    <jats:bold>Aim:<\/jats:bold>\n                    Hereditary angioedema (HAE) is a rare and chronic genetic condition. Lanadelumab and berotralstat, two plasma kallikrein inhibitors, have both been approved for long-term prophylaxis in patients with HAE; however, real-world data comparing costs and healthcare resource utilization (HCRU) are lacking.\n                    <jats:bold>Materials &amp; methods:<\/jats:bold>\n                    This retrospective study used administrative healthcare insurance claims data (Merative\u2122 MarketScan\n                    <jats:sup>\u00ae<\/jats:sup>\n                    Commercial, Medicare and Early View Research Databases; 1 July 2017\u201331 July 2023) to identify patients with HAE who initiated lanadelumab or berotralstat and were persistent for \u226518\u00a0months or 6\u00a0months, respectively. Sex, baseline healthcare costs and baseline number of on-demand treatment\/short-term prophylaxis medication claims were used to calculate covariate balancing propensity scores for inverse probability of treatment weighting. Following weighting, outcomes during the 6-month follow-up period in patients receiving berotralstat were compared with those during months 0\u20136, 7\u201312 and 13\u201318 in lanadelumab-treated patients.\n                    <jats:bold>Results:<\/jats:bold>\n                    Fifty-seven lanadelumab- and 32 berotralstat-treated patients were included. After weighting, more berotralstat-treated patients had an all-cause inpatient admission (berotralstat, 9.4%; lanadelumab, months 0\u20136, 4.0%, 7\u201312, 1.8%, months 13\u201318, 2.0%) and emergency room visit (berotralstat, 21.9%; lanadelumab, months 0\u20136, 14.0%, 7\u201312, 8.0%, months 13\u201318, 17.9%). Total HAE treatment costs were similar during months 0\u20136 (lanadelumab, $377,326 vs berotralstat, $373,010), but decreased in months 7\u201312 ($319,967) and 13\u201318 ($283,241) of lanadelumab. On-demand treatment\/short-term prophylaxis costs were lower for lanadelumab across the three follow-up periods than for berotralstat during months 0\u20136 (berotralstat, $60,451; lanadelumab, months 0\u20136, $46,336, months 7\u201312, $37,578, months 13\u201318, $23,968). The proportion of lanadelumab-treated patients who reduced dosing frequency was 24.8% during months 7\u201312 and 21.6% during months 13\u201318.\n                    <jats:bold>Conclusion:<\/jats:bold>\n                    Patients with HAE initiating lanadelumab versus berotralstat may require less on-demand and supportive HAE treatments and incur lower treatment-related and total healthcare costs. The ability to reduce lanadelumab dosing frequency after an attack-free period may be key in treatment selection, given the combination of cost savings and lower healthcare resource utilization.\n                  <\/jats:p>","DOI":"10.57264\/cer-2024-0205","type":"journal-article","created":{"date-parts":[[2025,2,20]],"date-time":"2025-02-20T06:11:01Z","timestamp":1740031861000},"source":"Crossref","is-referenced-by-count":3,"title":["Comparison of real-world healthcare resource utilization and costs among patients with hereditary angioedema on lanadelumab or berotralstat long-term prophylaxis"],"prefix":"10.57264","volume":"14","author":[{"given":"Nicole","family":"Princic","sequence":"first","affiliation":[{"name":"Merative, Ann Arbor, MI 48108, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kristin A","family":"Evans","sequence":"additional","affiliation":[{"name":"Merative, Ann Arbor, MI 48108, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chintal H","family":"Shah","sequence":"additional","affiliation":[{"name":"University of Maryland, Baltimore, MD 21201, USA"},{"name":"Takeda Pharmaceuticals USA, Inc., Lexington, MA 02421, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Krystal","family":"Sing","sequence":"additional","affiliation":[{"name":"Takeda Pharmaceuticals USA, Inc., Lexington, MA 02421, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Salom\u00e9","family":"Juethner","sequence":"additional","affiliation":[{"name":"Takeda Pharmaceuticals USA, Inc., Lexington, MA 02421, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bob G","family":"Schultz","sequence":"additional","affiliation":[{"name":"Takeda Pharmaceuticals USA, Inc., Lexington, MA 02421, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"36897","reference":[{"issue":"7","key":"e_1_3_8_3_2","doi-asserted-by":"crossref","first-page":"1961","DOI":"10.1111\/all.15214","article-title":"The international WAO\/EAACI guideline for the management of hereditary angioedema \u2013 the 2021 revision and update","volume":"77","author":"Maurer M","year":"2022","unstructured":"Maurer M, Magerl M, Betschel S The international WAO\/EAACI guideline for the management of hereditary angioedema \u2013 the 2021 revision and update. Allergy 77(7), 1961\u20131990 (2022).","journal-title":"Allergy"},{"issue":"14","key":"e_1_3_8_4_2","first-page":"S292","article-title":"Severity of hereditary angioedema, prevalence, and diagnostic considerations","volume":"24","author":"Bernstein JA","year":"2018","unstructured":"Bernstein JA. Severity of hereditary angioedema, prevalence, and diagnostic considerations. Am. J. Manag. Care 24(Suppl. 14), S292\u2013S298 (2018).","journal-title":"Am. J. Manag. Care"},{"issue":"6","key":"e_1_3_8_5_2","doi-asserted-by":"crossref","first-page":"100792","DOI":"10.1016\/j.waojou.2023.100792","article-title":"The multifactorial impact of receiving a hereditary angioedema diagnosis","volume":"16","author":"Raasch J","year":"2023","unstructured":"Raasch J, Glaum MC, O'Connor M. The multifactorial impact of receiving a hereditary angioedema diagnosis. World Allergy Organ J. 16(6), 100792 (2023).","journal-title":"World Allergy Organ J."},{"issue":"1","key":"e_1_3_8_6_2","doi-asserted-by":"crossref","first-page":"94","DOI":"10.1186\/s13023-021-01717-4","article-title":"Clinical characteristics and burden of illness in patients with hereditary angioedema: findings from a multinational patient survey","volume":"16","author":"Mendivil J","year":"2021","unstructured":"Mendivil J, Murphy R, de la Cruz M Clinical characteristics and burden of illness in patients with hereditary angioedema: findings from a multinational patient survey. Orphanet. J. Rare Dis. 16(1), 94 (2021).","journal-title":"Orphanet. J. Rare Dis."},{"issue":"1","key":"e_1_3_8_7_2","doi-asserted-by":"crossref","first-page":"S08","DOI":"10.2500\/aap.2020.41.200050","article-title":"Hereditary angioedema: epidemiology and burden of disease","volume":"41","author":"Lumry WR","year":"2020","unstructured":"Lumry WR, Settipane RA. Hereditary angioedema: epidemiology and burden of disease. Allergy Asthma Proc. 41(Suppl. 1), S08\u2013S13 (2020).","journal-title":"Allergy Asthma Proc."},{"issue":"1","key":"e_1_3_8_8_2","doi-asserted-by":"crossref","first-page":"132","DOI":"10.1016\/j.jaip.2020.08.046","article-title":"US HAEA Medical Advisory Board 2020 Guidelines for the Management of Hereditary Angioedema","volume":"9","author":"Busse PJ","year":"2021","unstructured":"Busse PJ, Christiansen SC, Riedl MA US HAEA Medical Advisory Board 2020 Guidelines for the Management of Hereditary Angioedema. J. Allergy Clin. Immunol. Pract. 9(1), 132\u201350.e3 (2021).","journal-title":"J. Allergy Clin. Immunol. Pract."},{"issue":"5","key":"e_1_3_8_9_2","doi-asserted-by":"crossref","first-page":"e12243","DOI":"10.1002\/clt2.12243","article-title":"Current challenges and future opportunities in patient-focused management of hereditary angioedema: a narrative review","volume":"13","author":"Grumach AS","year":"2023","unstructured":"Grumach AS, Gadir N, Kessel A, Yegin A, Martinez-Saguer I, Bernstein JA. Current challenges and future opportunities in patient-focused management of hereditary angioedema: a narrative review. Clin. Transl. Allergy 13(5), e12243 (2023).","journal-title":"Clin. Transl. Allergy"},{"key":"e_1_3_8_10_2","unstructured":"Takeda Pharmaceuticals USA Inc. TAKHZYRO\u2122 (lanadelumab-flyo) injection for subcutaneous use [prescribing information]. (2023). Available from: https:\/\/www.shirecontent.com\/PI\/PDFs\/TAKHZYRO_USA_ENG.pdf"},{"key":"e_1_3_8_11_2","unstructured":"BioCryst Pharmaceuticals. ORLADEYO\u2122 (berotralstat) capsules for oral use. (2020). Available from: https:\/\/www.accessdata.fda.gov\/drugsatfda_docs\/label\/2020\/214094s000lbl.pdf"},{"issue":"20","key":"e_1_3_8_12_2","doi-asserted-by":"crossref","first-page":"2108","DOI":"10.1001\/jama.2018.16773","article-title":"Effect of lanadelumab compared with placebo on prevention of hereditary angioedema attacks: a randomized clinical trial","volume":"320","author":"Banerji A","year":"2018","unstructured":"Banerji A, Riedl MA, Bernstein JA Effect of lanadelumab compared with placebo on prevention of hereditary angioedema attacks: a randomized clinical trial. J. Am. Med. Assoc. 320(20), 2108\u20132121 (2018).","journal-title":"J. Am. Med. Assoc."},{"issue":"3","key":"e_1_3_8_13_2","doi-asserted-by":"crossref","first-page":"979","DOI":"10.1111\/all.15011","article-title":"Long-term prevention of hereditary angioedema attacks with lanadelumab: the HELP OLE Study","volume":"77","author":"Banerji A","year":"2022","unstructured":"Banerji A, Bernstein JA, Johnston DT Long-term prevention of hereditary angioedema attacks with lanadelumab: the HELP OLE Study. Allergy 77(3), 979\u2013990 (2022).","journal-title":"Allergy"},{"issue":"1","key":"e_1_3_8_14_2","doi-asserted-by":"crossref","first-page":"164","DOI":"10.1016\/j.jaci.2020.10.015","article-title":"Oral once-daily berotralstat for the prevention of hereditary angioedema attacks: a randomized, double-blind, placebo-controlled phase III trial","volume":"148","author":"Zuraw B","year":"2021","unstructured":"Zuraw B, Lumry WR, Johnston DT Oral once-daily berotralstat for the prevention of hereditary angioedema attacks: a randomized, double-blind, placebo-controlled phase III trial. J. Allergy Clin. Immunol. Pract. 148(1), 164\u201372.e9 (2021).","journal-title":"J. Allergy Clin. Immunol. Pract."},{"key":"e_1_3_8_15_2","doi-asserted-by":"publisher","DOI":"10.57264\/cer-2022-0188"},{"key":"e_1_3_8_16_2","doi-asserted-by":"crossref","first-page":"22","DOI":"10.3389\/fmed.2018.00022","article-title":"Hereditary angioedema: the economics of treatment of an orphan disease","volume":"5","author":"Lumry WR","year":"2018","unstructured":"Lumry WR. Hereditary angioedema: the economics of treatment of an orphan disease. Front. Med. 5, 22 (2018).","journal-title":"Front. Med."},{"issue":"1","key":"e_1_3_8_17_2","doi-asserted-by":"crossref","first-page":"871","DOI":"10.1080\/13696998.2023.2232260","article-title":"Real-world changes in costs over time among patients in the United States with hereditary angioedema on long-term prophylaxis with lanadelumab","volume":"26","author":"Shah CH","year":"2023","unstructured":"Shah CH, Princic N, Evans KA, Schultz BG. Real-world changes in costs over time among patients in the United States with hereditary angioedema on long-term prophylaxis with lanadelumab. J. Med. Econ. 26(1), 871\u2013877 (2023).","journal-title":"J. Med. Econ."},{"issue":"1","key":"e_1_3_8_18_2","doi-asserted-by":"crossref","first-page":"243","DOI":"10.1111\/rssb.12027","article-title":"Covariate balancing propensity score","volume":"76","author":"Imai K","year":"2013","unstructured":"Imai K, Ratkovic M. Covariate balancing propensity score. J. R. Stat. Soc. Ser. B. Stat. Methodol. 76(1), 243\u2013263 (2013).","journal-title":"J. R. Stat. Soc. Ser. B. Stat. Methodol."},{"issue":"4","key":"e_1_3_8_19_2","doi-asserted-by":"crossref","first-page":"275","DOI":"10.2500\/aap.2023.44.230026","article-title":"Healthcare utilization of patients with hereditary angioedema treated with lanadelumab and subcutaneous C1-inhibitor concentrate","volume":"44","author":"Riedl MA","year":"2023","unstructured":"Riedl MA, Hinds DR, Prince PM Healthcare utilization of patients with hereditary angioedema treated with lanadelumab and subcutaneous C1-inhibitor concentrate. Allergy Asthma Proc. 44(4), 275\u2013282 (2023).","journal-title":"Allergy Asthma Proc."},{"issue":"10","key":"e_1_3_8_20_2","doi-asserted-by":"crossref","first-page":"3744","DOI":"10.1016\/j.jaip.2021.04.072","article-title":"Lanadelumab efficacy, safety, and injection interval extension in HAE: a real-life study","volume":"9","author":"Buttgereit T","year":"2021","unstructured":"Buttgereit T, Vera C, Weller K Lanadelumab efficacy, safety, and injection interval extension in HAE: a real-life study. J Allergy Clin. Immunol. Pract. 9(10), 3744\u20133751 (2021).","journal-title":"J Allergy Clin. Immunol. Pract."},{"key":"e_1_3_8_21_2","doi-asserted-by":"crossref","first-page":"1405317","DOI":"10.3389\/fimmu.2024.1405317","article-title":"The real life experience goes on: update after 4 years on the first cohort treated with lanadelumab at our center","volume":"15","author":"Buttgereit T","year":"2024","unstructured":"Buttgereit T, Vera Ayala C, Aykanat S The real life experience goes on: update after 4 years on the first cohort treated with lanadelumab at our center. Front. Immunol. 15, 1405317 (2024).","journal-title":"Front. Immunol."}],"container-title":["Journal of Comparative Effectiveness Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/becarispublishing.com\/doi\/pdf\/10.57264\/cer-2024-0205","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,3,21]],"date-time":"2025-03-21T07:40:46Z","timestamp":1742542846000},"score":1,"resource":{"primary":{"URL":"https:\/\/becarispublishing.com\/doi\/10.57264\/cer-2024-0205"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,4]]},"references-count":19,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2025,4]]}},"alternative-id":["10.57264\/cer-2024-0205"],"URL":"https:\/\/doi.org\/10.57264\/cer-2024-0205","relation":{},"ISSN":["2042-6305","2042-6313"],"issn-type":[{"value":"2042-6305","type":"print"},{"value":"2042-6313","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,4]]},"article-number":"e240205"}}