{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T11:04:54Z","timestamp":1790766294874,"version":"4.1.0"},"reference-count":86,"publisher":"Wiley","license":[{"start":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T00:00:00Z","timestamp":1790726400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"},{"start":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T00:00:00Z","timestamp":1790726400000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Muscle and Nerve"],"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Fatigue is a burdensome symptom for pediatric populations with neuromuscular disorders, often assessed using patient\u2010 and observer\u2010reported outcomes. The three aims of this systematic review are to: (1) explore, summarize and critically appraise primary studies that measure fatigue in pediatric neuromuscular disorders using patient\u2010 and observer\u2010reported outcomes; (2) compare definitions of fatigue and related terms presented within these studies; and (3) summarize evidence for validation of patient\u2010 and observer\u2010reported outcomes used in included studies. Six electronic databases were searched. Of 6801 studies identified, 64 met the inclusion criteria of measuring fatigue with a patient\u2010 and\/or observer\u2010reported outcome and investigated populations below 22\u2009years of age with neuromuscular disorders. Fatigue assessments included questionnaires (80%), interviews\/testimonials (17%), and single\u2010item measures (3%). To measure fatigue, 50% of studies used 1 questionnaire, 28% used 2 or more questionnaires, 6% used interviews alone, 3% used single\u2010item measures alone, and 13% used a combination of these methods. Quality appraisal indicated 80% of included studies had moderate\u2013high risk of bias. Of all included studies, 10 employed varying definitions of fatigue or related terms. Fatigue was commonly described as an overwhelming, multidimensional and whole\u2010body experience. Several questionnaires used to measure fatigue were developed to measure other constructs such as quality of life. Evidence for psychometric validation of all questionnaires was limited, and evidence for validation in neuromuscular disorders and pediatric populations was scarce. This review indicates a need for coherent and standardized fatigue definitions and measures while highlighting the scarcity of evidence for validation of frequently used questionnaires.<\/jats:p>","DOI":"10.1002\/mus.70416","type":"journal-article","created":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T10:30:38Z","timestamp":1790764238000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Systematic Review of Patient\u2010 and Observer\u2010Reported Outcomes of Fatigue in Pediatric Neuromuscular Disorders"],"prefix":"10.1002","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1631-2060","authenticated-orcid":false,"given":"Monica","family":"Marzouk","sequence":"first","affiliation":[{"name":"Faculty of Medicine and Health University of Sydney School of Health Sciences  Sydney New South Wales Australia"},{"name":"Sydney Children's Hospitals Network The Children's Hospital at Westmead  Westmead New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9863-1040","authenticated-orcid":false,"given":"Kayla M. D.","family":"Cornett","sequence":"additional","affiliation":[{"name":"Faculty of Medicine and Health University of Sydney School of Health Sciences  Sydney New South Wales Australia"},{"name":"Sydney Children's Hospitals Network The Children's Hospital at Westmead  Westmead New South Wales Australia"},{"name":"St. Jude Disability Prevention Program, Department of Epidemiology and Cancer Control St. Jude Children's Research Hospital  Memphis Tennessee USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4708-5879","authenticated-orcid":false,"given":"Gabrielle A.","family":"Donlevy","sequence":"additional","affiliation":[{"name":"Faculty of Medicine and Health University of Sydney School of Health Sciences  Sydney New South Wales Australia"},{"name":"Sydney Children's Hospitals Network The Children's Hospital at Westmead  Westmead New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-1220-2790","authenticated-orcid":false,"given":"Carl","family":"Young","sequence":"additional","affiliation":[{"name":"Paracelsus Medical University  Nuremberg Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7936-623X","authenticated-orcid":false,"given":"Joshua","family":"Burns","sequence":"additional","affiliation":[{"name":"St. Jude Disability Prevention Program, Department of Epidemiology and Cancer Control St. Jude Children's Research Hospital  Memphis Tennessee USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3160-6112","authenticated-orcid":false,"given":"Manoj P.","family":"Menezes","sequence":"additional","affiliation":[{"name":"Children's Hospital at Westmead Clinical School, Faculty of Medicine and Health University of Sydney  Sydney New South Wales Australia"},{"name":"TY Nelson Department of Neurology and Neurosurgery The Children's Hospital at Westmead  Sydney New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8126-3579","authenticated-orcid":false,"given":"Marnee J.","family":"McKay","sequence":"additional","affiliation":[{"name":"Faculty of Medicine and Health University of Sydney School of Health Sciences  Sydney New South Wales Australia"},{"name":"Sydney Children's Hospitals Network The Children's Hospital at Westmead  Westmead New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,9,30]]},"reference":[{"issue":"2","key":"e_1_2_11_2_1","doi-asserted-by":"crossref","first-page":"133","DOI":"10.1002\/mus.28305","article-title":"Fatigue and Fatigability in Spinal Muscular Atrophy; A Proposed Taxonomy to Enhance Assessment and Treatment","volume":"71","author":"Rodriguez\u2010Torres R.","year":"2025","journal-title":"Muscle & Nerve"},{"issue":"4","key":"e_1_2_11_3_1","doi-asserted-by":"crossref","first-page":"409","DOI":"10.1212\/WNL.0b013e31827f07be","article-title":"Fatigue and Fatigability in Neurologic Illnesses: Proposal for a Unified Taxonomy","volume":"80","author":"Kluger B. 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N.","year":"2024","journal-title":"European Journal of Paediatric Neurology"},{"issue":"1","key":"e_1_2_11_5_1","doi-asserted-by":"crossref","first-page":"78","DOI":"10.1186\/s41687-024-00758-0","article-title":"Identification of the Most Relevant Aspects of Spinal Muscular Atrophy (SMA) With Impact on the Quality of Life of SMA Patients and Their Caregivers: The PROfuture Project, A Qualitative Study","volume":"8","author":"Lemus M.","year":"2024","journal-title":"Journal of Patient\u2010Reported Outcomes"},{"issue":"4","key":"e_1_2_11_6_1","doi-asserted-by":"crossref","first-page":"429","DOI":"10.1177\/0883073812447681","article-title":"Severe Fatigue and Reduced Quality of Life in Children With Hereditary Motor and Sensory Neuropathy 1A","volume":"28","author":"Jagersma E.","year":"2013","journal-title":"Journal of Child Neurology"},{"issue":"1","key":"e_1_2_11_7_1","doi-asserted-by":"crossref","first-page":"13","DOI":"10.1186\/s13102-025-01057-x","article-title":"Health\u2010Related Quality of Life Associated With Fatigue, Physical Activity and Activity Pacing in Adults With Chronic Conditions","volume":"17","author":"Barakou I.","year":"2025","journal-title":"BMC Sports Science, Medicine and Rehabilitation"},{"issue":"2","key":"e_1_2_11_8_1","doi-asserted-by":"crossref","first-page":"105","DOI":"10.1111\/j.1744-6155.2007.00099.x","article-title":"The Relationship Between Fatigue and Quality of Life in Children With Chronic Health Problems: A Systematic Review","volume":"12","author":"Eddy L.","year":"2007","journal-title":"Journal for Specialists in Pediatric Nursing"},{"issue":"7","key":"e_1_2_11_9_1","doi-asserted-by":"crossref","first-page":"948","DOI":"10.1007\/s00415-006-0436-0","article-title":"Fatigue Is Part of the Burden of Neuromuscular Diseases","volume":"254","author":"Wokke J. 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