{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,2]],"date-time":"2026-05-02T01:55:21Z","timestamp":1777686921539,"version":"3.51.4"},"reference-count":22,"publisher":"SAGE Publications","issue":"2","license":[{"start":{"date-parts":[[2025,4,15]],"date-time":"2025-04-15T00:00:00Z","timestamp":1744675200000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"},{"start":{"date-parts":[[2025,4,15]],"date-time":"2025-04-15T00:00:00Z","timestamp":1744675200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"funder":[{"DOI":"10.13039\/100005725","name":"CHDI Foundation","doi-asserted-by":"publisher","award":["N\/A"],"award-info":[{"award-number":["N\/A"]}],"id":[{"id":"10.13039\/100005725","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Journal of Huntington's Disease"],"published-print":{"date-parts":[[2025,5]]},"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Current functional rating scales are not sensitive to the earliest functional changes in Huntington's disease (HD).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>The Functional Rating Scale 2.0 (FuRST 2.0) is a patient-reported outcome (PRO) measure designed to be sensitive to the initial functional changes in HD, specifically stage 2 and mild stage 3, as defined by the Huntington's Disease Integrated Staging System (HD-ISS).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We followed standard assessment development methodology to create a PRO. Study 1 consisted of a Delphi panel which analyzed data from focus groups comprised of people with HD and companions from 6 countries. This was followed by four rounds of cognitive interviews through which we evaluated respondents\u2019 comprehension of the instructions, understanding of question and response options, and comfort with the material. Informal advice from a regulatory agency was garnered throughout the process.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Concerns from the target population and regulators regarding instructions, questions, response options, and comfort with the material were addressed through modifications to the scale's wording and format.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>We developed the FuRST 2.0 official working document (OWD), the penultimate version of the scale, using focus groups, a Delphi panel, iterative rounds of cognitive interviewing, and informal regulatory advice. Its psychometric properties are being evaluated in the FOCUS-HD validation studies from which the final version of the scale will be derived. The FuRST 2.0 OWD is available for use.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1177\/18796397251323549","type":"journal-article","created":{"date-parts":[[2025,4,15]],"date-time":"2025-04-15T10:01:21Z","timestamp":1744711281000},"page":"171-178","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":5,"title":["Functional Rating Scale 2.0 (FuRST 2.0): A patient-reported outcome measure of function for Huntington\u2019s disease"],"prefix":"10.1177","volume":"14","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6545-7376","authenticated-orcid":false,"given":"Rebecca LM","family":"Fuller","sequence":"first","affiliation":[{"name":"CHDI Management, Inc., the company that manages the scientific activities of CHDI Foundation, Inc., Princeton, NJ, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0008-5146-7010","authenticated-orcid":false,"given":"Pua","family":"Feigenbaum","sequence":"additional","affiliation":[{"name":"CHDI Management, Inc., the company that manages the scientific activities of CHDI Foundation, Inc., Princeton, NJ, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nancy","family":"LaPelle","sequence":"additional","affiliation":[{"name":"Qualitative Research and Evaluation Consulting, Westborough, MA, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9972-955X","authenticated-orcid":false,"given":"Swati","family":"Sathe","sequence":"additional","affiliation":[{"name":"CHDI Management, Inc., the company that manages the scientific activities of CHDI Foundation, Inc., Princeton, NJ, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0007-5558-4188","authenticated-orcid":false,"given":"Prachi","family":"Dalal","sequence":"additional","affiliation":[{"name":"CHDI Management, Inc., the company that manages the scientific activities of CHDI Foundation, Inc., Princeton, NJ, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jatin G","family":"Vaidya","sequence":"additional","affiliation":[{"name":"Department of Psychiatry, University of Iowa, Iowa City, IA, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Neha","family":"Sinha","sequence":"additional","affiliation":[{"name":"CHDI Management, Inc., the company that manages the scientific activities of CHDI Foundation, Inc., Princeton, NJ, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Matthew","family":"Roch\u00e9","sequence":"additional","affiliation":[{"name":"CHDI Management, Inc., the company that manages the scientific activities of CHDI Foundation, Inc., Princeton, NJ, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8709-1384","authenticated-orcid":false,"given":"Cheryl J","family":"Fitzer-Attas","sequence":"additional","affiliation":[{"name":"ClinMed LLC, Monroe Township, NJ, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7052-9079","authenticated-orcid":false,"given":"Cristina","family":"Sampaio","sequence":"additional","affiliation":[{"name":"CHDI Management, Inc., the company that manages the scientific activities of CHDI Foundation, Inc., Princeton, NJ, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7905-9336","authenticated-orcid":false,"given":"Glenn T","family":"Stebbins","sequence":"additional","affiliation":[{"name":"Rush University Medical Center, Chicago, IL, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"179","published-online":{"date-parts":[[2025,4,15]]},"reference":[{"key":"e_1_3_5_2_2","doi-asserted-by":"publisher","DOI":"10.1186\/1750-1172-5-40"},{"key":"e_1_3_5_3_2","doi-asserted-by":"publisher","DOI":"10.1007\/978-1-4020-5614-7_1209"},{"key":"e_1_3_5_4_2","doi-asserted-by":"publisher","DOI":"10.1378\/chest.106.6.1645"},{"key":"e_1_3_5_5_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijnurstu.2003.09.004"},{"key":"e_1_3_5_6_2","doi-asserted-by":"publisher","DOI":"10.1002\/mds.870110204"},{"key":"e_1_3_5_7_2","doi-asserted-by":"publisher","DOI":"10.1002\/mdc3.12617"},{"key":"e_1_3_5_8_2","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.54.2.452"},{"key":"e_1_3_5_9_2","doi-asserted-by":"publisher","DOI":"10.1002\/mds.23337"},{"key":"e_1_3_5_10_2","article-title":"Assessment of day-to-day functioning in prodromal and early Huntington disease","volume":"3","author":"Vaccarino AL","year":"2011","unstructured":"Vaccarino AL, Sills T, Anderson KE, et al. 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