{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T05:14:50Z","timestamp":1787030090122,"version":"build-2736575974"},"reference-count":42,"publisher":"MDPI AG","issue":"9","license":[{"start":{"date-parts":[[2025,9,10]],"date-time":"2025-09-10T00:00:00Z","timestamp":1757462400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Office of Rare Diseases Research (ORDR) at the National Center for Advancing Clinical and Translational Studies","award":["U54 TR001456"],"award-info":[{"award-number":["U54 TR001456"]}]},{"name":"Office of Rare Diseases Research (ORDR) at the National Center for Advancing Clinical and Translational Studies","award":["U54 NS065701"],"award-info":[{"award-number":["U54 NS065701"]}]},{"name":"Office of Rare Diseases Research (ORDR) at the National Center for Advancing Clinical and Translational Studies","award":["U54 NS116025"],"award-info":[{"award-number":["U54 NS116025"]}]},{"name":"National Institute for Neurological Diseases and Stroke","award":["U54 TR001456"],"award-info":[{"award-number":["U54 TR001456"]}]},{"name":"National Institute for Neurological Diseases and Stroke","award":["U54 NS065701"],"award-info":[{"award-number":["U54 NS065701"]}]},{"name":"National Institute for Neurological Diseases and Stroke","award":["U54 NS116025"],"award-info":[{"award-number":["U54 NS116025"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Toxins"],"abstract":"<jats:p>Blepharospasm (BSP) is characterized by excessive orbicularis oculi muscle activity leading to abnormal blinking and involuntary eyelid closure. Botulinum neurotoxin (BoNT) injections are the main treatment for BSP, but they only partially and transiently relieve symptoms, leading to a waxing and waning therapeutic response. A patient-centered outcome (PCO) tool that measures BSP symptoms in a simple and efficient way could inform the development of better treatments. Using a stepwise modified Delphi approach, potential PCO items were first identified using the Dystonia Coalition Database with data from over 200 individuals with BSP who had provided responses to existing clinical assessment scales. These items were then analyzed for contribution to overall severity using a Random Forests approach, and redundant items were merged and revised in a series of iterative meetings with a specialist panel along with input from patient advocacy group representatives and focus groups. An online survey was conducted with 330 individuals with BSP to validate and verify the items\u2019 relevance. Finally, the specialist panel provided content validity ratio, which was repeated until it showed good agreement for relevance and clarity of all items. In the end, an easy-to-use PCO tool designed for smartphones and tablets containing 17 items covering three symptom domains (motor, disability, and psychosocial\/quality of life) was created. This novel PCO tool for BSP may be used to characterize the cyclical response that an individual patient experiences from BoNT treatments and provide a vital tool for future investigations of longer-acting BoNT preparations or adjunctive therapies.<\/jats:p>","DOI":"10.3390\/toxins17090455","type":"journal-article","created":{"date-parts":[[2025,9,10]],"date-time":"2025-09-10T08:43:54Z","timestamp":1757493834000},"page":"455","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Development of a Patient-Centered Outcome Tool for Blepharospasm: A Stepwise Modified Delphi Study"],"prefix":"10.3390","volume":"17","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0602-9942","authenticated-orcid":false,"given":"Brian D.","family":"Berman","sequence":"first","affiliation":[{"name":"Department of Neurology, Virginia Commonwealth University, Richmond, VA 23298, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fares","family":"Qeadan","sequence":"additional","affiliation":[{"name":"Department of Public Health Sciences, Loyola University Chicago, Chicago, IL 60153, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8676-4292","authenticated-orcid":false,"given":"Amanda D.","family":"Henderson","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, Wilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, MD 21287, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew R.","family":"Harrison","sequence":"additional","affiliation":[{"name":"Departments of Ophthalmology and Otolaryngology, University of Minnesota, Minneapolis, MN 55415, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Giovanni","family":"Defazio","sequence":"additional","affiliation":[{"name":"Department of Translational Biomedicine and Neuroscience, Aldo Moro University of Bari, 70124 Bari, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3180-6811","authenticated-orcid":false,"given":"Mark","family":"Hallett","sequence":"additional","affiliation":[{"name":"NINDS Intramural Program, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gamze","family":"Kilic-Berkmen","sequence":"additional","affiliation":[{"name":"Department of Neurology, Emory University School of Medicine, Atlanta, GA 30322, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura","family":"Wright","sequence":"additional","affiliation":[{"name":"Department of Neurology, Washington University School of Medicine, St. Louis, MO 63110, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0008-7510-7589","authenticated-orcid":false,"given":"Samantha","family":"Pentecost","sequence":"additional","affiliation":[{"name":"Department of Neurology, University of New Mexico Health Sciences Center, Albuquerque, NM 87106, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul","family":"Reyes","sequence":"additional","affiliation":[{"name":"Department of Neurology, University of New Mexico Health Sciences Center, Albuquerque, NM 87106, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna","family":"Tingin","sequence":"additional","affiliation":[{"name":"Department of Neurology, University of New Mexico Health Sciences Center, Albuquerque, NM 87106, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joseph","family":"Jankovic","sequence":"additional","affiliation":[{"name":"Parkinson\u2019s Disease Center and Movement Disorders Clinic, Department of Neurology, Baylor College of Medicine, Houston, TX 77030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jane","family":"Boyd","sequence":"additional","affiliation":[{"name":"Benign Essential Blepharospasm Research Foundation, Beaumont, TX 77726, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Charlene","family":"Hudgins","sequence":"additional","affiliation":[{"name":"Benign Essential Blepharospasm Research Foundation, Beaumont, TX 77726, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Janet","family":"Hieshetter","sequence":"additional","affiliation":[{"name":"Dystonia Medical Research Foundation, Chicago, IL 60601, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joel S.","family":"Perlmutter","sequence":"additional","affiliation":[{"name":"Department of Neurology, Washington University School of Medicine, St. Louis, MO 63110, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1531-3969","authenticated-orcid":false,"given":"Hyder A.","family":"Jinnah","sequence":"additional","affiliation":[{"name":"Department of Neurology, Emory University School of Medicine, Atlanta, GA 30322, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarah","family":"Pirio Richardson","sequence":"additional","affiliation":[{"name":"Department of Neurology, University of New Mexico Health Sciences Center, Albuquerque, NM 87106, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2025,9,10]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"109","DOI":"10.1016\/j.parkreldis.2021.09.004","article-title":"Diagnostic Criteria for Blepharospasm: A Multicenter International Study","volume":"91","author":"Defazio","year":"2021","journal-title":"Park. 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