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Multiple sclerosis (MS) is multifactorial in nature, with both genetic and environmental factors contributing to disease risk. In this umbrella review, we aimed to summarize various non-HLA genes that have been investigated for their association with MS risk in meta-analyses (MAs) of observational studies.Medline, Embase, Epistemonikos, and Cochrane Database of Systematic Reviews were searched for MAs up to July 2021. Evidence of association was ranked according to established criteria as follows: convincing, highly suggestive, suggestive, weak, or not significant.From 1,533 publications, 85 full-text articles were evaluated for eligibility, and 15 articles were selected that described 114 genetic associations. No associations were supported by convincing evidence. One association between the rs2104286 (A vs G contrast) polymorphism of the interleukin 2RA (IL2RA) gene and increased MS susceptibility was initially supported by highly suggestive evidence (OR 1.17; 95% CI 1.10-1.25) but it was downgraded to suggestive in the sensitivity and subgroup analyses. We also found six associations supported by suggestive evidence in the main analysis.The findings of this study imply that the cytokines genes interleukin IL2RA and IL7R may play a role in the development of MS. Data from more and better-designed studies are needed to establish robust evidence.

Background High and ever-increasing costs of Multiple Sclerosis (MS) disease-modifying therapies (DMTs) have impaired patient access to DMTs in the US. Patients’ willingness-to-pay (WTPs) for DMTs were recently examined, but their influencers were not determined. Thus, the objective of this study was to examine factors influencing patients’ WTPs for DMTs for multiple sclerosis (MS). Methods Data were obtained from a previous survey of 1,200 US patients with MS on their preferences and WTPs for DMTs. Patients’ characteristics (i.e., age, gender, race, marital status, education, employment status, comorbidity, health status, and health insurance) and their MS experiences (i.e., number of years with MS, MS type, number of relapses, fatigue, mood-change, MS symptom, and DMTs experience) were investigated as influencing factors. Patient's WTP for a DMT was obtained from a direct question in the survey. A two-part model was estimated using logistic regression and generalized linear regression. Results Responses from 480 patients were analyzed. Their average age was 53 years old. Most of them were female (79%), white (97%), and married (71%). Approximately 61% of them had a four-year college degree or lower, 54% were either unemployed, retired, or students, 59% were enrolled in private insurance, 81% had at least one comorbid condition, and 73% considered themselves having good or better health status. Approximately 44% had at least one relapse in two years, 89% experienced fatigue, 37% experienced mood-change, and on average had MS for more than 13 years. The majority of them had relapsing-remitting MS (66%), considered themselves to have some levels of disability for MS (78%), and had used or were currently using DMTs (97%). The average WTP for a DMT was $579 per month. Patients with professional degrees, or with one or more comorbid conditions were more likely willing to pay for a DMT. Patients who were white, had a professional degree, or were in fair or better health status were willing to pay a significantly higher amount for a DMT. Patients, who were female, were employed, did not have private insurance, had a higher number of MS experience years, or who experienced mood change were willing to pay significantly less amount for a DMT. Conclusion Various patients’ characteristics and MS experiences, including gender, race, education, employment, health insurance, comorbidity, health status, DMT experience, and mood change influenced patients’ WTPs for a DMT.

Background While disease-modifying therapies (DMTs) for multiple sclerosis (MS) treatments are costly, patient valuation of DMTs has not been examined. The objective of this study was to examine patients' preferences and willingness-to-pay (WTP) for DMTs. Methods Six attributes (i.e., number of relapses, percentage of disability progression, percentage of severe adverse events, route of administration, frequency of administration, and out-of-pocket cost) and their levels were used to develop a discrete choice experiment questionnaire. Each questionnaire comprised seven choice sets and each choice set contained two hypothetical DMTs and an opt-out alternative. A total of 1,200 U.S. patients with MS were asked to choose a DMT option or opt-out in each choice set. Multinomial logit model was used to determine relative preferences of each attribute. WTPs for all attributes and DMTs were calculated. Results A total of 508 patients were analyzed. Patients preferred DMTs with lower relapse rate, lower disability progression, lower severe adverse event, lower frequency of administration, and lower cost. In addition, they preferred oral DMTs. They were willing to pay $2,768, $289, $292, and $76 a month in exchange for every 1-time decrease in the number of relapses in two years, every 1% decrease in disability progression in two years, every 1% decrease in severe adverse events, and every 1-time decrease in the frequency of administration per month, respectively. The patients were willing to pay, in relation to market prices, between $7,020 and $134,934 per year for all DMTs, but interferon beta-1a SC. Conclusions Patients with MS considered relapse rate, disability progression, severe adverse events, route of administration, frequency of administration, and out-of-pocket cost, when they chose DMTs. Their WTPs for DMTs varied widely.