The Economic and Humanistic Burden of Pediatric-Onset Multiple Sclerosis

**Background:** 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. **Objectives:** To conduct a literature r...

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Main Authors: Nupur Greene, Lita Araujo, Cynthia Campos, Hannah Dalglish, Sarah Gibbs, Irina Yermilov
Format: Article
Language:English
Published: Columbia Data Analytics, LLC 2022-10-01
Series:Journal of Health Economics and Outcomes Research
Online Access:https://doi.org/10.36469/001c.37992
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author Nupur Greene
Lita Araujo
Cynthia Campos
Hannah Dalglish
Sarah Gibbs
Irina Yermilov
author_facet Nupur Greene
Lita Araujo
Cynthia Campos
Hannah Dalglish
Sarah Gibbs
Irina Yermilov
author_sort Nupur Greene
collection DOAJ
description **Background:** 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. **Objectives:** 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. **Methods:** 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. **Results:** 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 $38 543 (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™ 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. **Discussion:** 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. **Conclusions:** Healthcare resource utilization and costs are high in this population, and patients report reduced QOL and significant fatigue compared with healthy children and adolescents.
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spelling doaj-art-b4c03337508a45eb99ff5ec3d6a17c8c2025-02-10T16:13:05ZengColumbia Data Analytics, LLCJournal of Health Economics and Outcomes Research2327-22362022-10-0192The Economic and Humanistic Burden of Pediatric-Onset Multiple SclerosisNupur GreeneLita AraujoCynthia CamposHannah DalglishSarah GibbsIrina Yermilov**Background:** 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. **Objectives:** 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. **Methods:** 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. **Results:** 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 $38 543 (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™ 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. **Discussion:** 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. **Conclusions:** Healthcare resource utilization and costs are high in this population, and patients report reduced QOL and significant fatigue compared with healthy children and adolescents.https://doi.org/10.36469/001c.37992
spellingShingle Nupur Greene
Lita Araujo
Cynthia Campos
Hannah Dalglish
Sarah Gibbs
Irina Yermilov
The Economic and Humanistic Burden of Pediatric-Onset Multiple Sclerosis
Journal of Health Economics and Outcomes Research
title The Economic and Humanistic Burden of Pediatric-Onset Multiple Sclerosis
title_full The Economic and Humanistic Burden of Pediatric-Onset Multiple Sclerosis
title_fullStr The Economic and Humanistic Burden of Pediatric-Onset Multiple Sclerosis
title_full_unstemmed The Economic and Humanistic Burden of Pediatric-Onset Multiple Sclerosis
title_short The Economic and Humanistic Burden of Pediatric-Onset Multiple Sclerosis
title_sort economic and humanistic burden of pediatric onset multiple sclerosis
url https://doi.org/10.36469/001c.37992
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