{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,11,28]],"date-time":"2025-11-28T12:33:29Z","timestamp":1764333209132,"version":"3.43.0"},"reference-count":71,"publisher":"The Journal of Health Economics and Outcomes Research","issue":"2","license":[{"start":{"date-parts":[[2022,10,18]],"date-time":"2022-10-18T00:00:00Z","timestamp":1666051200000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["jheor.org"],"crossmark-restriction":true},"short-container-title":[],"abstract":"<jats:sec id=\"background\"> <jats:title>Background<\/jats:title> <jats:p>Multiple sclerosis (MS) is a chronic inflammatory autoimmune disease of the central nervous system. Pediatric-onset MS (POMS), defined as onset of MS before 18 years of age, is estimated to account for 2% to 5% of the MS population worldwide.<\/jats:p> <\/jats:sec> <jats:sec id=\"objectives\"> <jats:title>Objectives<\/jats:title> <jats:p>To conduct a literature review focused on the healthcare resource utilization and cost as well as quality-of-life (QOL) outcomes among patients with POMS.<\/jats:p> <\/jats:sec> <jats:sec id=\"methods\"> <jats:title>Methods<\/jats:title> <jats:p>We conducted a systematic literature review of English-language studies published after September 2010 in MEDLINE and Embase to describe the global economic healthcare resource utilization and costs and humanistic (QOL) burden in patients with POMS.<\/jats:p> <\/jats:sec> <jats:sec id=\"results\"> <jats:title>Results<\/jats:title> <jats:p>We found 11 studies that reported on healthcare resource utilization, cost, or insurance coverage and 36 studies that reported on QOL outcomes in patients with POMS. Patients with POMS had higher rates of primary care visits (1.41 [1.29-1.54]), hospital visits (10.74 [8.95-12.90]), and admissions (rate ratio, 4.27 [2.92-6.25];OR, 15.2 [12.0-19.1]) compared with healthy controls. Mean per-patient costs in the United States were $5907 across all settings per year of follow-up between 2002 and 2012; mean costs per hospital stay were $38\u202f543 (in 2015 USD) between 2004 and 2013. Three studies reported psychosocial scores between 71.59 and 79.7, and 8 studies reported physical health scores between 74.62 to 82.75 using the Pediatric Quality of Life Measurement Model (PedsQLTM). Twelve studies used the PedsQL\u2122 Multidimensional Fatigue Scale. Mean scores on the self-reported general fatigue scale ranged from 63.15 to 78.5. Quality-of-life scores were lower than those of healthy controls.<\/jats:p> <\/jats:sec> <jats:sec id=\"discussion\"> <jats:title>Discussion<\/jats:title> <jats:p>Our review presents a uniquely broad and recent overview of the global economic and humanistic burden of patients with POMS. Additional research on healthcare resource utilization and cost would provide a more robust understanding of the economic burden in this population.<\/jats:p> <\/jats:sec> <jats:sec id=\"conclusions\"> <jats:title>Conclusions<\/jats:title> <jats:p>Healthcare resource utilization and costs are high in this population, and patients report reduced QOL and significant fatigue compared with healthy children and adolescents.<\/jats:p> <\/jats:sec>","DOI":"10.36469\/001c.37992","type":"journal-article","created":{"date-parts":[[2022,10,18]],"date-time":"2022-10-18T19:51:41Z","timestamp":1666122701000},"update-policy":"https:\/\/doi.org\/10.36469\/00001","source":"Crossref","is-referenced-by-count":2,"title":["The Economic and Humanistic Burden of Pediatric-Onset Multiple Sclerosis"],"prefix":"10.36469","volume":"9","author":[{"given":"Nupur","family":"Greene","sequence":"first","affiliation":[{"name":"Health Economics & Value Assessment, Sanofi, Cambridge, Massachusetts"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lita","family":"Araujo","sequence":"additional","affiliation":[{"name":"Health Economics & Value Assessment, Sanofi, Cambridge, Massachusetts"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Cynthia","family":"Campos","sequence":"additional","affiliation":[{"name":"PHAR (Partnership for Health Analytic Research), LLC, Beverly Hills, California"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hannah","family":"Dalglish","sequence":"additional","affiliation":[{"name":"PHAR (Partnership for Health Analytic Research), LLC, Beverly Hills, California"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sarah","family":"Gibbs","sequence":"additional","affiliation":[{"name":"PHAR (Partnership for Health Analytic Research), LLC, Beverly Hills, California"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Irina","family":"Yermilov","sequence":"additional","affiliation":[{"name":"PHAR (Partnership for Health Analytic Research), LLC, Beverly Hills, California"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"22273","published-online":{"date-parts":[[2022,10,18]]},"reference":[{"issue":"3","key":"ref1","doi-asserted-by":"publisher","first-page":"229","DOI":"10.1111\/dmcn.12964","article-title":"Understanding fatigue in paediatric multiple sclerosis: a systematic review of clinical and psychosocial factors","volume":"58","author":"Susan Carroll","year":"2016","journal-title":"Developmental Medicine &amp; Child Neurology","ISSN":"https:\/\/id.crossref.org\/issn\/0012-1622","issn-type":"print"},{"issue":"10","key":"ref2","doi-asserted-by":"publisher","first-page":"50","DOI":"10.1007\/s11940-019-0592-z","article-title":"Newer treatment approaches in pediatric-onset multiple sclerosis","volume":"21","author":"Gabrielle Macaron","year":"2019","journal-title":"Current Treatment Options in Neurology","ISSN":"https:\/\/id.crossref.org\/issn\/1092-8480","issn-type":"print"},{"issue":"6","key":"ref3","doi-asserted-by":"publisher","first-page":"73","DOI":"10.3390\/children6060073","article-title":"Long-term treatment strategies of pediatric multiple sclerosis, including the use of disease modifying therapies","volume":"6","author":"Mary Rensel","year":"2019","journal-title":"Children","ISSN":"https:\/\/id.crossref.org\/issn\/2227-9067","issn-type":"print"},{"issue":"4","key":"ref4","doi-asserted-by":"publisher","first-page":"321","DOI":"10.1080\/14737167.2017.1358617","article-title":"Estimate of the cost of multiple sclerosis in Spain by literature review","volume":"17","author":"Oscar Fern\u00e1ndez","year":"2017","journal-title":"Expert Review of Pharmacoeconomics &amp; Outcomes Research","ISSN":"https:\/\/id.crossref.org\/issn\/1473-7167","issn-type":"print"},{"issue":"5","key":"ref5","doi-asserted-by":"publisher","first-page":"639","DOI":"10.3111\/13696998.2013.778268","article-title":"The cost burden of multiple sclerosis in the United States: a systematic review of the literature","volume":"16","author":"Gabriel Adelman","year":"2013","journal-title":"Journal of Medical Economics","ISSN":"https:\/\/id.crossref.org\/issn\/1369-6998","issn-type":"print"},{"key":"ref6","article-title":"Disease progression in pediatric multiple sclerosis: disparities between physical and neurocognitive outcomes","volume":"9","author":"E.A. 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