Greene Nupur, Araujo Lita, Campos Cynthia, Dalglish Hannah, Gibbs Sarah, Yermilov Irina
Health Economics & Value Assessment, Sanofi, Cambridge, Massachusetts.
PHAR (Partnership for Health Analytic Research), LLC, Beverly Hills, California.
J Health Econ Outcomes Res. 2022 Oct 18;9(2):103-114. doi: 10.36469/001c.37992. eCollection 2022.
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. 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. 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. 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. 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. Healthcare resource utilization and costs are high in this population, and patients report reduced QOL and significant fatigue compared with healthy children and adolescents.
多发性硬化症(MS)是一种中枢神经系统的慢性炎症性自身免疫性疾病。儿童期发病的多发性硬化症(POMS)定义为18岁之前发病的MS,据估计占全球MS患者群体的2%至5%。为了进行一项聚焦于POMS患者医疗资源利用、成本以及生活质量(QOL)结果的文献综述。我们对2010年9月之后发表在MEDLINE和Embase上的英文研究进行了系统的文献综述,以描述POMS患者的全球经济医疗资源利用、成本以及人文(QOL)负担。我们发现11项研究报告了医疗资源利用、成本或保险覆盖情况,36项研究报告了POMS患者的QOL结果。与健康对照组相比,POMS患者的初级保健就诊率更高(1.41[1.29 - 1.54])、医院就诊率更高(10.74[8.95 - 12.90])以及住院率更高(率比,4.27[2.92 - 6.25];比值比,15.2[12.0 - 19.1])。在2002年至2012年的随访期间,美国每位患者每年在所有环境下的平均成本为5907美元;在2004年至2013年期间,每次住院的平均成本为38543美元(2015年美元)。三项研究报告的心理社会得分在71.59至79.7之间,八项研究使用儿童生活质量测量模型(PedsQLTM)报告的身体健康得分在74.62至82.75之间。十二项研究使用了PedsQL™多维疲劳量表。自我报告的一般疲劳量表的平均得分在63.15至78.5之间。生活质量得分低于健康对照组。我们的综述对POMS患者的全球经济和人文负担进行了独特而广泛且最新的概述。对医疗资源利用和成本的进一步研究将更深入地了解该人群的经济负担。该人群的医疗资源利用和成本很高,并且与健康儿童和青少年相比,患者报告生活质量下降且疲劳显著。