{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,2]],"date-time":"2026-10-02T13:41:48Z","timestamp":1790948508553,"version":"4.1.0"},"reference-count":28,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2025,8,6]],"date-time":"2025-08-06T00:00:00Z","timestamp":1754438400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2025,8,6]],"date-time":"2025-08-06T00:00:00Z","timestamp":1754438400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100004339","name":"Sanofi","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100004339","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Orphanet J Rare Dis"],"abstract":"<jats:title>Abstract<\/jats:title>\n          <jats:sec>\n            <jats:title>Background<\/jats:title>\n            <jats:p>Warm Autoimmune Hemolytic Anemia (wAIHA) is a rare form of anemia caused by premature destruction of red cells. This qualitative study aimed to understand the patient experience with wAIHA and provide evidence of FACIT-Fatigue scale validity by interviewing adult wAIHA patients along with examining patients\u2019 definitions of meaningful change for item 1 of the FACIT Fatigue.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Methods<\/jats:title>\n            <jats:p>Hybrid Concept Elicitation (CE) and Cognitive Debriefing (CD) interviews, lasting approximately 75\u00a0min, were conducted with US-based adults diagnosed with wAIHA. The CE part of the interview focused on understanding the patient's experiences with wAIHA, particularly the signs\/symptoms, and impacts. The CD part evaluated patient\u2019s feedback on clarity, relevance, and comprehensibility of the FACIT-Fatigue, and examined their interpretation of meaningful change for item 1.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Results<\/jats:title>\n            <jats:p>Nineteen adult wAIHA patients (mean age 49.7\u00a0years) from the US were interviewed, 31 signs\/symptoms were identified with fatigue being the most common (reported by all patients, 100%) and bothersome. In addition, many patients experienced shortness of breath (95%) and skin\/eye color change (74%). Other common symptoms included headaches (63%), dark\/brown urine (63%), heart palpitations (63%), and dizziness (53%). The study also found that wAIHA can have negative impact on patients\u2019 emotional functioning, physical functioning, daily activities, social and professional life. The most frequently reported impacts included feeling anxious or scared (79%), difficulty climbing stairs (74%), needing help from others (74%), and reduced physical strength\/mobility (74%). Patients also frequently reported feelings of anger\/frustration (68%), needing frequent rests (68%), difficulty walking (68%), feeling worrisome (53%), and impacts on social activities (53%). A novel conceptual disease model was developed, highlighting the associations between symptoms and their impacts on patients\u2019 lives. Patients indicated the FACIT-Fatigue was clear, relevant, and appropriate for measuring their wAIHA related fatigue. A 1-point score change in the item 1 was considered meaningful for both improvement and worsening of their fatigue by most patients.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Conclusions<\/jats:title>\n            <jats:p>The study offers insight into the patient experience with wAIHA, detailing common symptoms and impacts on daily life as part of wAIHA conceptual disease model. It also supports the content validity of the FACIT-Fatigue instrument for assessing fatigue in wAIHA patients.<\/jats:p>\n          <\/jats:sec>","DOI":"10.1186\/s13023-025-03767-4","type":"journal-article","created":{"date-parts":[[2025,8,6]],"date-time":"2025-08-06T06:33:58Z","timestamp":1754462038000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Exploring patients\u2019 experiences with wAIHA and the content validity of the FACIT-fatigue: a qualitative interview study"],"prefix":"10.1186","volume":"20","author":[{"given":"Anubha","family":"Shukla","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Owen","family":"Cooper","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karen","family":"Jones","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Quentin A.","family":"Hill","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Keith","family":"McCrae","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bruno","family":"Fattizzo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael","family":"Kostikas","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6153-0828","authenticated-orcid":false,"given":"Marieke","family":"Krol","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stephanie","family":"Bascle","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sylvie","family":"Bozzi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ahmed","family":"Daak","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joshua","family":"Maher","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2025,8,6]]},"reference":[{"issue":"7","key":"3767_CR1","doi-asserted-by":"publisher","first-page":"647","DOI":"10.1056\/NEJMcp1900554","volume":"381","author":"RA Brodsky","year":"2019","unstructured":"Brodsky RA. 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The research protocol, interview guide, and all patient communication templates were reviewed and approved by WIRB-Copernicus Group (WCG) Institutional Review Board (IRB) prior to the study commencing (#20225959) and all patients gave written informed consent to participate.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Not applicable.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"Joshua Maher, Stephanie Bascle, Ahmed Daak, and Sylvie Bozzi are employees and may hold stock and\/or stock options in Sanofi. Anubha Shukla, Owen Cooper, Michael Kostikas and Marieke Krol are employees of IQVIA who provided research services for Sanofi for this study. Karen Jones is an employee of wAIHA Warriors. wAIHA Warriors received remuneration from IQVIA for assisting in identifying and recruiting patients with wAIHA to participate in this study. Quentin Hill provides consultancy to Alpine, Amgen, Argenx, Gliknik, Incyte, Immunovant, Janssen, Novartis, ReAlta, Sanofi and Sobi, and is speaker honoraria at Grifols and Novartis.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"403"}}