{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,31]],"date-time":"2026-08-31T03:14:16Z","timestamp":1788146056871,"version":"build-2803163510"},"reference-count":53,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2026,6,19]],"date-time":"2026-06-19T00:00:00Z","timestamp":1781827200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2026,8,31]],"date-time":"2026-08-31T00:00:00Z","timestamp":1788134400000},"content-version":"vor","delay-in-days":73,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100013669","name":"Ionis Pharmaceuticals","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100013669","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["J Patient Rep Outcomes"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Psychometric validation of the Angioedema Quality of Life Questionnaire (AE-QoL) was conducted to determine its suitability for assessing health-related quality of life (HRQoL) in patients with hereditary angioedema (HAE).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methodology<\/jats:title>\n                    <jats:p>Data were analyzed from 90 participants with HAE in OASIS-HAE (NCT05139810), a randomized, double-blind, placebo-controlled phase 3 study of donidalorsen (DAWNZERA\u2122). Psychometric validation, assessed by post hoc analyses on scores at Baseline and Week 24 visits, included internal consistency reliability, structural and construct validity (convergent, criterion, and known-groups validity), and responsiveness.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Confirmatory factor analysis supported the 17-item AE-QoL measurement model (comparative fit index [CFI], Tucker-Lewis index [TLI], and standardized root mean square residual [SRMR] met thresholds). Internal consistency reliability was strong for the total, functioning, fatigue\/mood, and fears\/shame domains (Cronbach\u2019s \u237a: 0.81\u20130.97; McDonald\u2019s \u2375: 0.86\u20130.98), but weaker for the 2-item nutrition domain (Cronbach\u2019s \u237a: 0.65\u20130.75; McDonald\u2019s \u2375: 0.64\u20130.75). At Week 24, AE-QoL total, functioning, and fears\/shame scores showed convergent validity with the 28-day Angioedema Activity Score (AAS-28) (n\n                      <jats:italic>r<\/jats:italic>\n                      \u2009=\u20090.51, 0.73, and 0.51, respectively) and with the Angioedema Control Test (AECT) (\n                      <jats:italic>r<\/jats:italic>\n                      =-0.77, -0.89, and \u2212\u20090.70, respectively). Criterion validity was supported by moderate-to-strong correlations with HAE attack rate at Week 24 (total:\n                      <jats:italic>r<\/jats:italic>\n                      \u2009=\u20090.65; domains:\n                      <jats:italic>r<\/jats:italic>\n                      \u2009=\u20090.55\u20130.78, excluding fatigue\/mood). Known-groups validity was demonstrated by significant differences in AE-QoL scores between patients with poor- versus well-controlled disease based on AECT score at Baseline and Week 24 (all\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.05, Cohen\u2019s\n                      <jats:italic>d<\/jats:italic>\n                      effect size [ES] range: 0.63\u20133.65). AE-QoL total, functioning, and fears\/shame scores were sensitive to changes in disease activity, with correlations\u2019 absolute values for changes in score from Baseline to Week 24 exceeding 0.30 for AAS-28 and 0.39 for AECT, excluding fatigue\/mood and nutrition domains. AE-QoL change scores for total, functioning, fears\/shame, and nutrition domains effectively differentiated between subgroups with varying reductions in HAE attack frequency (all\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.02, ES range: 0.34\u20131.42).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>The AE-QoL demonstrated strong reliability, validity, and ability to detect change, supporting its use as an important patient-reported outcome measure for evaluating HRQoL in patients with HAE.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s41687-026-01128-8","type":"journal-article","created":{"date-parts":[[2026,6,19]],"date-time":"2026-06-19T07:46:01Z","timestamp":1781855161000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Psychometric validation of the Angioedema Quality of Life Questionnaire (AE-QoL) for hereditary angioedema"],"prefix":"10.1186","volume":"10","author":[{"given":"Alexandra J.","family":"Feld","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martha","family":"Bayliss","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jakob B.","family":"Bjorner","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Regina","family":"Rendas-Baum","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cary","family":"Thurm","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aaron","family":"Yarlas","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2026,6,19]]},"reference":[{"issue":"6","key":"1128_CR1","doi-asserted-by":"publisher","first-page":"100792","DOI":"10.1016\/j.waojou.2023.100792","volume":"16","author":"J Raasch","year":"2023","unstructured":"Raasch J, Glaum MC, O\u2019Connor M (2023) The multifactorial impact of receiving a hereditary angioedema diagnosis. World Allergy Organ J 16(6):100792. https:\/\/doi.org\/10.1016\/j.waojou.2023.100792","journal-title":"World Allergy Organ J"},{"issue":"8","key":"1128_CR2","doi-asserted-by":"publisher","first-page":"2450","DOI":"10.1016\/j.jaip.2023.01.023","volume":"11","author":"MA Riedl","year":"2023","unstructured":"Riedl MA, Danese M, Danese S, Ulloa J, Maetzel A, Audhya PK (2023) Hereditary angioedema with normal C1 inhibitor: US survey of prevalence and provider practice patterns. J Allergy Clin Immunol Pract 11(8):2450\u20132456e6. https:\/\/doi.org\/10.1016\/j.jaip.2023.01.023","journal-title":"J Allergy Clin Immunol Pract"},{"issue":"1","key":"1128_CR3","first-page":"48","volume":"121","author":"H Zafra","year":"2022","unstructured":"Zafra H (2022) Hereditary angioedema: A review. Wis Med J 121(1):48\u201353","journal-title":"Wis Med J"},{"issue":"3","key":"1128_CR4","doi-asserted-by":"publisher","first-page":"267","DOI":"10.1016\/j.amjmed.2005.09.064","volume":"119","author":"K Bork","year":"2006","unstructured":"Bork K, Meng G, Staubach P, Hardt J (2006) Hereditary angioedema: New findings concerning symptoms, affected organs, and course. Am J Med 119(3):267\u2013274. https:\/\/doi.org\/10.1016\/j.amjmed.2005.09.064","journal-title":"Am J Med"},{"issue":"6","key":"1128_CR5","doi-asserted-by":"publisher","first-page":"600","DOI":"10.1016\/j.anai.2020.02.018","volume":"124","author":"A Banerji","year":"2020","unstructured":"Banerji A, Davis KH, Brown TM, Hollis K, Hunter SM, Long J, Jain G, Devercelli G (2020) Patient-reported burden of hereditary angioedema: Findings from a patient survey in the United States. Ann Allergy Asthma Immunol 124(6):600\u2013607. https:\/\/doi.org\/10.1016\/j.anai.2020.02.018","journal-title":"Ann Allergy Asthma Immunol"},{"key":"1128_CR6","doi-asserted-by":"publisher","first-page":"101339","DOI":"10.1016\/j.ienj.2023.101339","volume":"71","author":"AE Burton","year":"2023","unstructured":"Burton AE, Lindsay-Wiles I, Herron D, Owen A, Elliott J, Metcalfe A, Diwakar L (2023) Hereditary angioedema patient experiences of medication use and emergency care. Int Emerg Nurs 71:101339. https:\/\/doi.org\/10.1016\/j.ienj.2023.101339","journal-title":"Int Emerg Nurs"},{"issue":"3","key":"1128_CR7","doi-asserted-by":"publisher","first-page":"100758","DOI":"10.1016\/j.waojou.2023.100758","volume":"16","author":"HJ Chong-Neto","year":"2023","unstructured":"Chong-Neto HJ (2023) A narrative review of recent literature of the quality of life in hereditary angioedema patients. World Allergy Organ J 16(3):100758. https:\/\/doi.org\/10.1016\/j.waojou.2023.100758","journal-title":"World Allergy Organ J"},{"issue":"1","key":"1128_CR8","doi-asserted-by":"publisher","first-page":"1","DOI":"10.18176\/jiaci.0653","volume":"31","author":"T Caballero","year":"2021","unstructured":"Caballero T (2021) Treatment of hereditary angioedema. J Investig Allergol Clin Immunol 31(1):1\u201316. https:\/\/doi.org\/10.18176\/jiaci.0653","journal-title":"J Investig Allergol Clin Immunol"},{"issue":"4","key":"1128_CR9","doi-asserted-by":"publisher","first-page":"371","DOI":"10.1016\/j.anai.2013.05.028","volume":"112","author":"AS Fouche","year":"2014","unstructured":"Fouche AS, Saunders EFH, Craig T (2014) Depression and anxiety in patients with hereditary angioedema. Ann Allergy Asthma Immunol 112(4):371\u2013375. https:\/\/doi.org\/10.1016\/j.anai.2013.05.028","journal-title":"Ann Allergy Asthma Immunol"},{"issue":"5","key":"1128_CR10","doi-asserted-by":"publisher","first-page":"407","DOI":"10.2500\/aap.2010.31.3394","volume":"31","author":"WR Lumry","year":"2010","unstructured":"Lumry WR, Castaldo AJ, Vernon MK, Blaustein MB, Wilson DA, Horn PT (2010) The humanistic burden of hereditary angioedema: Impact on health-related quality of life, productivity, and depression. Allergy Asthma Proc 31(5):407\u2013414. https:\/\/doi.org\/10.2500\/aap.2010.31.3394","journal-title":"Allergy Asthma Proc"},{"issue":"1","key":"1128_CR11","doi-asserted-by":"publisher","first-page":"40","DOI":"10.1186\/s13223-021-00537-2","volume":"17","author":"K Bork","year":"2021","unstructured":"Bork K, Anderson JT, Caballero T, Craig T, Johnston DT, Li HH, Longhurst HJ, Radojicic C, Riedl MA (2021) Assessment and management of disease burden and quality of life in patients with hereditary angioedema: A consensus report. Allergy Asthma Clin Immunol 17(1):40. https:\/\/doi.org\/10.1186\/s13223-021-00537-2","journal-title":"Allergy Asthma Clin Immunol"},{"issue":"1","key":"1128_CR12","doi-asserted-by":"publisher","first-page":"1","DOI":"10.5415\/apallergy.0000000000000172","volume":"15","author":"R Sharma","year":"2025","unstructured":"Sharma R, Basu S, Tyagi R, Siniah S, Barman P, Sil A, Machhua S, Chawla S, Pandiarajan V, Pilania RK, Sharma S, Dhaliwal M, Suri D, Rawat A, Kumaran SM, Dogra S, Singh S, Jindal AK (2025) Quality of life in patients with hereditary angioedema correlates with angioedema control: Our experience at Chandigarh, India. Asia Pac Allergy 15(1):1\u20136. https:\/\/doi.org\/10.5415\/apallergy.0000000000000172","journal-title":"Asia Pac Allergy"},{"key":"1128_CR13","doi-asserted-by":"publisher","unstructured":"Lumry WR, Zuraw B, Cicardi M, Craig T, Anderson J, Banerji A, Bernstein JA, Caballero T, Farkas H, Gower RG, Keith PK, Levy DS, Li HH, Magerl M, Manning M, Riedl MA, Lawo JP, Prusty S, Machnig T, on behalf of the COMPACT Investigators (2021) Long-term health-related quality of life in patients treated with subcutaneous C1-inhibitor replacement therapy for the prevention of hereditary angioedema attacks: findings from the COMPACT open-label extension study. Orphanet Journal of Rare Diseases.16(1). https:\/\/doi.org\/10.1186\/s13023-020-01658-4","DOI":"10.1186\/s13023-020-01658-4"},{"key":"1128_CR14","doi-asserted-by":"publisher","unstructured":"Anderson J, Levy DS, Lumry WR, Koochaki P, Lanar S, Li HH (2021) Letting the patients speak: An in-depth qualitative research-based investigation of factors relevant to health-related quality of life in real-world patients with hereditary angioedema using subcutaneous C1 inhibitor replacement therapy. Allergy, Asthma & Clinical Immunology. 17(1). https:\/\/doi.org\/10.1186\/s13223-021-00550-5","DOI":"10.1186\/s13223-021-00550-5"},{"key":"1128_CR15","doi-asserted-by":"publisher","unstructured":"Hews-Girard J, Goodyear MD (2021) Psychosocial burden of type 1 and 2 hereditary angioedema: a single-center Canadian cohort study. Allergy, Asthma & Clinical Immunology.17(1). https:\/\/doi.org\/10.1186\/s13223-021-00563-0","DOI":"10.1186\/s13223-021-00563-0"},{"issue":"1","key":"1128_CR16","doi-asserted-by":"publisher","first-page":"50","DOI":"10.1007\/s12016-021-08850-9","volume":"61","author":"Z Balla","year":"2021","unstructured":"Balla Z, Ign\u00e1cz B, Varga L, K\u0151halmi KV, Farkas H (2021) How Angioedema Quality of Life Questionnaire can help physicians in treating C1-inhibitor deficiency patients? Clin Rev Allergy Immunol 61(1):50\u201359. https:\/\/doi.org\/10.1007\/s12016-021-08850-9","journal-title":"Clin Rev Allergy Immunol"},{"key":"1128_CR17","doi-asserted-by":"publisher","unstructured":"Mendivil J, Murphy R, De La Cruz M, Janssen E, Boysen HB, Jain G, Ayg\u00f6ren-P\u00fcrs\u00fcn E, Hirji I, Devercelli G (2021) Clinical characteristics and burden of illness in patients with hereditary angioedema: findings from a multinational patient survey. Orphanet Journal of Rare Diseases.16(1). https:\/\/doi.org\/10.1186\/s13023-021-01717-4","DOI":"10.1186\/s13023-021-01717-4"},{"issue":"6","key":"1128_CR18","doi-asserted-by":"publisher","first-page":"703","DOI":"10.1016\/j.anai.2024.07.018","volume":"133","author":"AJ Castaldo","year":"2024","unstructured":"Castaldo AJ, Siersb\u00e6k N, Corcoran D, Selva CN, Jervelund C, Christiansen SC, Riedl MA, Zuraw BL, Busse PJ (2024) Development and validation of a US quality of life instrument for hereditary angioedema due to C1 inhibitor deficiency. Ann Allergy Asthma Immunol 133(6):703\u2013711e6. https:\/\/doi.org\/10.1016\/j.anai.2024.07.018","journal-title":"Ann Allergy Asthma Immunol"},{"issue":"1","key":"1128_CR19","doi-asserted-by":"publisher","first-page":"82","DOI":"10.1186\/1477-7525-10-82","volume":"10","author":"N Prior","year":"2012","unstructured":"Prior N, Remor E, G\u00f3mez-Traseira C, L\u00f3pez-Serrano C, Caba\u00f1as R, Contreras J, Campos \u00c1, Cardona V, Cimbollek S, Gonz\u00e1lez-Quevedo T, Guilarte M, De Rojas DH, Marcos C, Rubio M, Tejedor-Alonso M, Caballero T (2012) Development of a disease-specific quality of life questionnaire for adult patients with hereditary angioedema due to C1 inhibitor deficiency (HAE-QoL): Spanish multi-centre research project. Health Qual Life Outcomes 10(1):82. https:\/\/doi.org\/10.1186\/1477-7525-10-82","journal-title":"Health Qual Life Outcomes"},{"issue":"10","key":"1128_CR20","doi-asserted-by":"publisher","first-page":"1289","DOI":"10.1111\/all.12007","volume":"67","author":"K Weller","year":"2012","unstructured":"Weller K, Groffik A, Magerl M, Tohme N, Martus P, Krause K, Metz M, Staubach P, Maurer M (2012) Development and construct validation of the angioedema quality of life questionnaire. Allergy 67(10):1289\u20131298. https:\/\/doi.org\/10.1111\/all.12007","journal-title":"Allergy"},{"key":"1128_CR21","doi-asserted-by":"publisher","unstructured":"Vanya M, Watt M, Shahraz S, Kosmas CE, Rhoten S, Costa-Cabral S, Mendivil J, Devercelli G, Weller K (2023) Content validation and psychometric evaluation of the Angioedema Quality of Life Questionnaire for hereditary angioedema. Journal of Patient-Reported Outcomes. 7(1). https:\/\/doi.org\/10.1186\/s41687-023-00576-w","DOI":"10.1186\/s41687-023-00576-w"},{"key":"1128_CR22","doi-asserted-by":"publisher","unstructured":"Broderick L, Foster A, Waldman LT, Bordone L, Yarlas A (2025) The adolescent experience of hereditary angioedema: a qualitative study of disease burden and treatment experience. Orphanet Journal of Rare Diseases. 20(1). https:\/\/doi.org\/10.1186\/s13023-025-03539-0","DOI":"10.1186\/s13023-025-03539-0"},{"key":"1128_CR23","doi-asserted-by":"publisher","unstructured":"Broderick L, Foster A, Waldman LT, Jacobs K, Bordone L, Yarlas A (2025) Content validation of the Angioedema Quality of Life Questionnaire (AE-QoL) in a population of adult and adolescent patients with hereditary angioedema (HAE). Journal of Patient-Reported Outcomes. 9(1). https:\/\/doi.org\/10.1186\/s41687-025-00876-3","DOI":"10.1186\/s41687-025-00876-3"},{"key":"1128_CR24","unstructured":"U.S. Food and Drug Administration (2024) Patient-focused drug development: selecting, developing, or modifying fit-for-purpose clinical outcome assessments [Internet]. Available from: (accessed 22 May 2026) https:\/\/www.fda.gov\/. Accessed 22 May 2026"},{"issue":"1","key":"1128_CR25","doi-asserted-by":"publisher","first-page":"21","DOI":"10.1056\/NEJMoa2402478","volume":"391","author":"MA Riedl","year":"2024","unstructured":"Riedl MA, Tachdjian R, Lumry WR, Craig T, Karakaya G, Gelincik A, Stobiecki M, Jacobs JS, Gokmen NM, Reshef A, Gompels MM, Manning ME, Bordone L, Newman KB, Treadwell S, Wang S, Yarlas A, Cohn DM (2024) Efficacy and safety of donidalorsen for hereditary angioedema. N Engl J Med 391(1):21\u201331. https:\/\/doi.org\/10.1056\/NEJMoa2402478","journal-title":"N Engl J Med"},{"issue":"1","key":"1128_CR26","doi-asserted-by":"publisher","first-page":"160","DOI":"10.1186\/s12955-019-1229-3","volume":"17","author":"K Kulthanan","year":"2019","unstructured":"Kulthanan K, Chularojanamontri L, Rujitharanawong C, Weerasubpong P, Maurer M, Weller K (2019) Angioedema quality of life questionnaire (AE-QoL)\u2014Interpretability and sensitivity to change. Health Qual Life Outcomes 17(1):160. https:\/\/doi.org\/10.1186\/s12955-019-1229-3","journal-title":"Health Qual Life Outcomes"},{"issue":"9","key":"1128_CR27","doi-asserted-by":"publisher","first-page":"1185","DOI":"10.1111\/all.12209","volume":"68","author":"K Weller","year":"2013","unstructured":"Weller K, Groffik A, Magerl M, Tohme N, Martus P, Krause K, Metz M, Staubach P, Maurer M (2013) Development, validation, and initial results of the Angioedema Activity Score. Allergy 68(9):1185\u20131192. https:\/\/doi.org\/10.1111\/all.12209","journal-title":"Allergy"},{"issue":"5","key":"1128_CR28","doi-asserted-by":"publisher","first-page":"1165","DOI":"10.1111\/all.14144","volume":"75","author":"K Weller","year":"2020","unstructured":"Weller K, Donoso T, Magerl M, Ayg\u00f6ren-P\u00fcrs\u00fcn E, Staubach P, Martinez-Saguer I, Hawro T, Altrichter S, Krause K, Siebenhaar F, Metz M, Zuberbier T, Freier D, Maurer M (2020) Development of the Angioedema Control Test - A patient-reported outcome measure that assesses disease control in patients with recurrent angioedema. Allergy 75(5):1165\u20131177. https:\/\/doi.org\/10.1111\/all.14144","journal-title":"Allergy"},{"issue":"6","key":"1128_CR29","doi-asserted-by":"publisher","first-page":"2050","DOI":"10.1016\/j.jaip.2020.02.038","volume":"8","author":"K Weller","year":"2020","unstructured":"Weller K, Donoso T, Magerl M, Ayg\u00f6ren-P\u00fcrs\u00fcn E, Staubach P, Martinez-Saguer I, Hawro T, Altrichter S, Krause K, Siebenhaar F, Metz M, Zuberbier T, Freier D, Maurer M (2020) Validation of the Angioedema Control Test (AECT)\u2014A patient-reported outcome instrument for assessing angioedema control. J Allergy Clin Immunol 8(6):2050\u20132057e4. https:\/\/doi.org\/10.1016\/j.jaip.2020.02.038","journal-title":"J Allergy Clin Immunol"},{"issue":"1","key":"1128_CR30","doi-asserted-by":"publisher","first-page":"4","DOI":"10.1177\/0146621606289485","volume":"31","author":"RD Gibbons","year":"2007","unstructured":"Gibbons RD, Bock RD, Hedeker D, Weiss DJ, Segawa E, Bhaumik DK, Kupfer DJ, Frank E, Grochocinski VJ, Stover A (2007) Full-information item bifactor analysis of graded response data. Appl Psychol Meas 31(1):4\u201319. https:\/\/doi.org\/10.1177\/0146621606289485","journal-title":"Appl Psychol Meas"},{"issue":"1","key":"1128_CR31","doi-asserted-by":"publisher","first-page":"72","DOI":"10.1037\/a0032138","volume":"19","author":"GJ Geldhof","year":"2014","unstructured":"Geldhof GJ, Preacher KJ, Zyphur MJ (2014) Reliability estimation in a multilevel confirmatory factor analysis framework. Psychol Methods 19(1):72\u201391. https:\/\/doi.org\/10.1037\/a0032138","journal-title":"Psychol Methods"},{"issue":"2","key":"1128_CR32","doi-asserted-by":"publisher","first-page":"137","DOI":"10.1037\/met0000045","volume":"21","author":"A Rodriguez","year":"2016","unstructured":"Rodriguez A, Reise SP, Haviland MG (2016) Evaluating bifactor models: Calculating and interpreting statistical indices. Psychol Methods 21(2):137\u2013150. https:\/\/doi.org\/10.1037\/met0000045","journal-title":"Psychol Methods"},{"issue":"1","key":"1128_CR33","doi-asserted-by":"publisher","first-page":"53","DOI":"10.21427\/D7CF7R","volume":"6","author":"D Hooper","year":"2008","unstructured":"Hooper D, Coughlan J, Mullen MR (2008) Structural equation modelling: Guidelines for determining model fit. Electron J Bus Res Methods 6(1):53\u201360. https:\/\/doi.org\/10.21427\/D7CF7R","journal-title":"Electron J Bus Res Methods"},{"issue":"4","key":"1128_CR34","doi-asserted-by":"publisher","first-page":"462","DOI":"10.1177\/0049124108314720","volume":"36","author":"C Feinian","year":"2008","unstructured":"Feinian C, Curran PJ, Bollen KA, Kirby J, Paxton P (2008) An empirical evaluation of the use of fixed cutoff points in RMSEA test statistic in structural equation models. Sociol Methods Res 36(4):462\u2013494. https:\/\/doi.org\/10.1177\/0049124108314720","journal-title":"Sociol Methods Res"},{"issue":"2","key":"1128_CR35","doi-asserted-by":"publisher","first-page":"230","DOI":"10.1177\/0049124192021002005","volume":"21","author":"MW Browne","year":"1992","unstructured":"Browne MW, Cudeck R (1992) Alternative ways of assessing model fit. Sociol Methods Res 21(2):230\u2013258. https:\/\/doi.org\/10.1177\/0049124192021002005","journal-title":"Sociol Methods Res"},{"key":"1128_CR36","unstructured":"Muth\u00e9n & Muth\u00e9n (n.d.) Mplus. Los Angeles, CA"},{"key":"1128_CR37","unstructured":"Brown TA (2006) Confirmatory factor analysis for applied research. New York (NY): Guilford Press"},{"issue":"1","key":"1128_CR38","doi-asserted-by":"publisher","first-page":"34","DOI":"10.1016\/j.jclinepi.2006.03.012","volume":"60","author":"CB Terwee","year":"2007","unstructured":"Terwee CB, Bot SDM, De Boer MR, Van Der Windt DAWM, Knol DL, Dekker J, Bouter LM, De Vet HCW (2007) Quality criteria were proposed for measurement properties of health status questionnaires. J Clin Epidemiol 60(1):34\u201342. https:\/\/doi.org\/10.1016\/j.jclinepi.2006.03.012","journal-title":"J Clin Epidemiol"},{"key":"1128_CR39","unstructured":"Nunnally JC, Bernstein IH (2008) Psychometric theory. 3rd ed. New York (NY): McGraw-Hill"},{"key":"1128_CR40","doi-asserted-by":"crossref","unstructured":"Cappelleri JC, Zou KH, Bushmakin AG, Alvir JM, Alemayehu D, Symonds T (2013) Patient-reported outcomes: measurement, implementation and interpretation. Boca Raton (FL): CRC Press","DOI":"10.1201\/b16139"},{"issue":"2","key":"1128_CR41","doi-asserted-by":"publisher","first-page":"81","DOI":"10.1037\/h0046016","volume":"56","author":"DT Campbell","year":"1959","unstructured":"Campbell DT, Fiske DW (1959) Convergent and discriminant validation by the multitrait-multimethod matrix. Psychol Bull 56(2):81\u2013105. https:\/\/doi.org\/10.1037\/h0046016","journal-title":"Psychol Bull"},{"key":"1128_CR42","doi-asserted-by":"publisher","unstructured":"Cohen J (1988) Statistical power analysis for the behavioral sciences (2nd ed.). Lawrence Erlbaum Associates. https:\/\/doi.org\/10.4324\/9780203771587","DOI":"10.4324\/9780203771587"},{"issue":"2","key":"1128_CR43","doi-asserted-by":"publisher","first-page":"102","DOI":"10.1016\/j.jclinepi.2007.03.012","volume":"61","author":"D Revicki","year":"2008","unstructured":"Revicki D, Hays RD, Cella D, Sloan J (2008) Recommended methods for determining responsiveness and minimally important differences for patient-reported outcomes. J Clin Epidemiol 61(2):102\u2013109. https:\/\/doi.org\/10.1016\/j.jclinepi.2007.03.012","journal-title":"J Clin Epidemiol"},{"issue":"4","key":"1128_CR44","doi-asserted-by":"publisher","first-page":"1188","DOI":"10.1111\/all.14680","volume":"76","author":"WR Lumry","year":"2021","unstructured":"Lumry WR, Weller K, Magerl M, Banerji A, Longhurst HJ, Riedl MA, Lewis HB, Lu P, Devercelli G, Jain G, Maurer M, the HELP Study Investigators (2021) Impact of lanadelumab on health-related quality of life in patients with hereditary angioedema in the HELP study. Allergy 76(4):1188\u20131198. https:\/\/doi.org\/10.1111\/all.14680","journal-title":"Allergy"},{"key":"1128_CR45","doi-asserted-by":"publisher","unstructured":"Zuraw BL, Lumry WR, Johnston DT, Ayg\u00f6ren-P\u00fcrs\u00fcn E, Banerji A, Bernstein JA, Christiansen SC, Jacobs JS, Sitz KV, Gower RG, Gagnon R, Wedner HJ, Kinaciyan T, Hakl R, Hanzl\u00edkov\u00e1 J, Anderson JT, McNeil DL, Fritz SB, Yang WH, et al (2021) Oral once-daily berotralstat for the prevention of hereditary angioedema attacks: a randomized, double-blind, placebo-controlled phase 3 trial. Journal of Allergy and Clinical Immunology.148(1):164\u2013172.e9. https:\/\/doi.org\/10.1016\/j.jaci.2020.10.015","DOI":"10.1016\/j.jaci.2020.10.015"},{"key":"1128_CR46","doi-asserted-by":"publisher","unstructured":"Craig TJ, Reshef A, Li HH, Jacobs JS, Bernstein JA, Farkas H, Yang WH, Stroes ESG, Ohsawa I, Tachdjian R, Manning ME, Lumry WR, Saguer IM, Ayg\u00f6ren-P\u00fcrs\u00fcn E, Ritchie B, Sussman GL, Anderson J, Kawahata K, Suzuki Y, et al (2023) Efficacy and safety of garadacimab, a factor XIIa inhibitor for hereditary angioedema prevention (VANGUARD): a global, multicentre, randomised, double-blind, placebo-controlled, phase 3 trial. The Lancet. 401(10382):1079\u20131090. https:\/\/doi.org\/10.1016\/S0140-6736(23)00350-1","DOI":"10.1016\/S0140-6736(23)00350-1"},{"issue":"8","key":"1128_CR47","doi-asserted-by":"publisher","first-page":"1203","DOI":"10.1111\/all.12900","volume":"71","author":"K Weller","year":"2016","unstructured":"Weller K, Magerl M, Peveling-Oberhag A, Martus P, Staubach P, Maurer M (2016) The Angioedema Quality of Life Questionnaire (AE \u2010QoL) \u2013 Assessment of sensitivity to change and minimal clinically important difference. Allergy 71(8):1203\u20131209. https:\/\/doi.org\/10.1111\/all.12900","journal-title":"Allergy"},{"issue":"7","key":"1128_CR48","doi-asserted-by":"publisher","first-page":"100554","DOI":"10.1016\/j.waojou.2021.100554","volume":"14","author":"PK Can","year":"2021","unstructured":"Can PK, Degi\u0307rmentepe EN, Etikan P, Kizilta\u00e7 K, Gelincik A, Demir S, Buyukozturk S, Ha\u015fal E, B\u00fclb\u00fcl Ba\u015fkan E, Aydin \u00d6, Maurer M, Weller K, Kocaturk E (2021) Assessment of disease activity and quality of life in patients with recurrent bradykinin-mediated versus mast cell-mediated angioedema. World Allergy Organ J 14(7):100554. https:\/\/doi.org\/10.1016\/j.waojou.2021.100554","journal-title":"World Allergy Organ J"},{"issue":"4","key":"1128_CR49","doi-asserted-by":"publisher","first-page":"471","DOI":"10.1016\/j.alit.2021.04.006","volume":"70","author":"S Morioke","year":"2021","unstructured":"Morioke S, Takahagi S, Kawano R, Fukunaga A, Harada S, Ohsawa I, Masuda K, Irifuku R, Yokobayashi H, Kameyoshi Y, Tanaka A, Tamari S, Hide M (2021) A validation study of the Japanese version of the Angioedema Activity Score (AAS) and the Angioedema Quality of Life Questionnaire (AE-QoL). Allergol Int 70(4):471\u2013479. https:\/\/doi.org\/10.1016\/j.alit.2021.04.006","journal-title":"Allergol Int"},{"issue":"1","key":"1128_CR50","doi-asserted-by":"publisher","first-page":"15","DOI":"10.1007\/s12016-021-08839-4","volume":"61","author":"I Leibovich-Nassi","year":"2021","unstructured":"Leibovich-Nassi I, Reshef A (2021) The enigma of prodromes in hereditary angioedema (HAE). Clin Rev Allergy Immunol 61(1):15\u201328. https:\/\/doi.org\/10.1007\/s12016-021-08839-4","journal-title":"Clin Rev Allergy Immunol"},{"issue":"6","key":"1128_CR51","doi-asserted-by":"publisher","first-page":"505","DOI":"10.1111\/cea.14632","volume":"55","author":"HWF Mak","year":"2025","unstructured":"Mak HWF, Wong JCY, Chiang V, Lam DLY, Li PH (2025) From anxiety to work productivity and activity impairment: The mediating role of fatigue in hereditary angioedema. Clin Exp Allergy 55(6):505\u2013507. https:\/\/doi.org\/10.1111\/cea.14632","journal-title":"Clin Exp Allergy"},{"issue":"3","key":"1128_CR52","doi-asserted-by":"publisher","first-page":"S4","DOI":"10.2500\/aap.2021.42.210025","volume":"42","author":"C Radojicic","year":"2021","unstructured":"Radojicic C, Riedl MA, Craig TJ, Best JM, Rosselli J, Hahn R, Banerji A (2021) Patient perspectives on the treatment burden of injectable medication for hereditary angioedema. Allergy Asthma Proc 42(3):S4\u2013S10. https:\/\/doi.org\/10.2500\/aap.2021.42.210025","journal-title":"Allergy Asthma Proc"},{"issue":"1","key":"1128_CR53","doi-asserted-by":"publisher","first-page":"75","DOI":"10.5114\/ada.2024.144488","volume":"42","author":"MSB Durmaz","year":"2025","unstructured":"Durmaz MSB, Sevimli N (2025) Attack frequency and associated factors in hereditaryangioedema patients: A single-centre experience. Postepy Dermatol Alergol 42(1):75\u201382. https:\/\/doi.org\/10.5114\/ada.2024.144488","journal-title":"Postepy Dermatol Alergol"}],"container-title":["Journal of Patient-Reported Outcomes"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s41687-026-01128-8","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s41687-026-01128-8.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s41687-026-01128-8.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,31]],"date-time":"2026-08-31T02:29:33Z","timestamp":1788143373000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1186\/s41687-026-01128-8"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,6,19]]},"references-count":53,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2026,12]]}},"alternative-id":["1128"],"URL":"https:\/\/doi.org\/10.1186\/s41687-026-01128-8","relation":{},"ISSN":["2509-8020"],"issn-type":[{"value":"2509-8020","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,6,19]]},"assertion":[{"value":"2 April 2026","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"14 June 2026","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"19 June 2026","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"The OASIS-HAE multicenter international study was performed in line with the principles of the Declaration of Helsinki and Good Practice Guidelines. Study protocols were reviewed and approved by an independent ethics committee or institutional review board at each participating clinical study site\/country. No single ethics approval number exists. Trial registration numbers are: ClinicalTrials.gov: NCT05139810 (OASIS HAE) and EudraCT: 2021 002571 19.","order":1,"name":"Ethics","label":"Ethics approval","group":{"name":"EthicsHeading","label":"<b>Decleration<\/b>"}},{"value":"Not applicable.","order":2,"name":"Ethics","label":"Consent for publication","group":{"name":"EthicsHeading","label":"<b>Decleration<\/b>"}},{"value":"Written informed consent was obtained from all individual participants included in the study.","order":3,"name":"Ethics","label":"Consent to participate","group":{"name":"EthicsHeading","label":"<b>Decleration<\/b>"}},{"value":"AJF, MB, JBB, RRB, and CT are current or former employees of IQVIA, which received funding from Ionis to conduct this research. AY is an employee of and holds shares and options in Ionis Pharmaceuticals, Inc., which funded this research.","order":4,"name":"Ethics","label":"Competing interests","group":{"name":"EthicsHeading","label":"<b>Decleration<\/b>"}}],"article-number":"159"}}