The Economic and Humanistic Burden of Pediatric-Onset Multiple Sclerosis

Abstract

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.

Rituximab treatment in pediatric‐onset multiple sclerosis

“…Pediatric-onset MS (POMS) is now recognized as a non-benign inflammatory demyelinating disease associated with a high risk of early physical and cognitive impairment [3,4], fatigue [6] and psychiatric comorbidities [29], leading to a significant impact on families and high healthcare resource utilization [6]. Therefore, there is increasing consensus on the need for early initiation of highly effective therapies [30,31] in young population.…”

“…Yet at disease onset brain volumes are smaller compared to age‐matched controls and brain growth is occurring at slower rates [5]. Healthcare resource utilization and costs are high in this population and patients report a reduction in quality of life and significant fatigue compared to age‐matched controls [6].…”

Multiple Sclerosis—Related Dietary and Nutritional Issues: An Updated Scoping Review with a Focus on Pediatrics

“…The median age of children with POMS is 12 years (range: 1.6–17). It is not clear whether the recently reported increased incidence may be real or more likely dependent on better diagnostic methods [ 2 , 153 , 154 , 155 , 156 ]. As outlined before, at any rate, data in POMS remain scarce mainly due to small sample size [ 1 , 157 ].…”

“…Consequently, healthcare resource utilization and costs are high in this population compared with healthy children and adolescents. Quite common cognitive impairment requiring specific management, decrease in QOL, and an increase in economic burden in POMS have been shown to have profound impacts not only on patients but also on their families [ 154 , 155 ]. Still, an early onset of MS seems to be somewhat protective regarding QOL [ 165 ], maybe due to better resilience resources in youth [ 154 , 155 , 166 ].…”

“…Quite common cognitive impairment requiring specific management, decrease in QOL, and an increase in economic burden in POMS have been shown to have profound impacts not only on patients but also on their families [ 154 , 155 ]. Still, an early onset of MS seems to be somewhat protective regarding QOL [ 165 ], maybe due to better resilience resources in youth [ 154 , 155 , 166 ]. Efforts towards a better understanding of possible specific mechanisms that trigger POMS and improving inherent therapies and prevention appear, therefore, to be necessary in young and pre-pubertal children [ 162 , 167 , 168 ].…”