Epidemiology of pediatric multiple sclerosis: A systematic literature review and meta-analysis

Yan, Kevin; Balijepalli, Chakrapani; Desai, Kamal; Gullapalli, Lakshmi; Druyts, Eric

doi:10.1016/j.msard.2020.102260

Cited by 52 publications

(57 citation statements)

References 46 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…POMS is defined as the manifestation of MS symptoms under the age of 16 or 18 [ 58 ]. According to our analysis, the frequency of FMS in POMS was higher than AOMS (Fig.…”

Section: Discussionmentioning

confidence: 99%

The global prevalence of familial multiple sclerosis: an updated systematic review and meta-analysis

2021

View full text Add to dashboard Cite

Background Considering that many recent studies have reported the prevalence of familial multiple sclerosis (FMS), we performed an updated meta-analysis of the worldwide prevalence of FMS by the addition of recent publications. Methods A search in PubMed, Scopus, the ISI Web of Science, and Google Scholar was undertaken up to 20 December 2020. The inclusion criteria were based on the CoCoPop approach (condition, context, and population). Meta-analysis of the qualified studies was conducted by comprehensive meta-analysis ver. 2 software. Results The pooled prevalence of MS in relatives of 16,179 FMS cases was estimated to be 11.8% (95% CI: 10.7–13) based on a random-effects model. The pooled mean age of disease onset in adult probands was calculated to be 28.7 years (95% CI: 27.2 ± 30.2). Regarding 13 studies that reported the data of FMS in pediatrics (n = 877) and adults (n = 6636), the FMS prevalence in pediatrics and adults was 15.5% (95% CI: 13.8–17.4) and 10.8% (95% CI: 8.1–14.2), respectively. The prevalence of FMS in affected males (n = 5243) and females (n = 11,503) was calculated to be 13.7% (95% CI: 10.1–18.2) and 15.4% (95% CI: 10.3–22.4), respectively. The odds ratio of male/female in FMS cases was not statistically significant (OR = 0.9; 95% CI: 0.6–1.2, P = 0.55). Subgroup analysis demonstrated a significant difference in the prevalence of FMS between the geographical areas (P = 0.007). The meta-regression model indicated that the prevalence of FMS is lower with higher latitude and higher MS prevalence (P < 0.001). In contrast, meta-regression based on prevalence day was not statistically significant (P = 0.29). Conclusions The prevalence of FMS is higher in the pediatric group than that of adults, distinct between geographical areas, and diminishes with the increment of MS prevalence and latitude. Also, the symptoms initiate relatively at younger ages in the FMS cases. Interestingly, our analysis unveiled that FMS is not more prevalent in men than women and the risk of MS development in relatives is not higher when the affected proband is male.

show abstract

“…POMS is defined as the manifestation of MS symptoms under the age of 16 or 18 [ 58 ]. According to our analysis, the frequency of FMS in POMS was higher than AOMS (Fig.…”

Section: Discussionmentioning

confidence: 99%

The global prevalence of familial multiple sclerosis: an updated systematic review and meta-analysis

2021

View full text Add to dashboard Cite

show abstract

“…The first attack of CNS demyelination in children (termed, acquired demyelinating syndromes (ADS)) represents the initial clinical presentation of multiple sclerosis (MS), anti-myelin oligodendrocyte glycoprotein associated disease (MOGAD), anti-AQP4 associated neuromyelitis optica spectrum disorder (AQP4-NMOSD), or can occur as a monophasic illness [A: in adults or children, or both?]. Global incidence of pediatric ADS is estimated as 0.87 (95% CI: 0.35-1.40) per 100,000 children per year 1 . Approximately 20% of children with ADS will be confirmed to have MS 2,3 , although these pediatric-onset MS patients represent fewer than 5% of all MS patients 4 .…”

Section: Introductionmentioning

confidence: 99%

Paediatric multiple sclerosis and antibody-associated demyelination: clinical, imaging, and biological considerations for diagnosis and care

Fadda

Armangué

Hacohen

et al. 2021

The Lancet Neurology

View full text Add to dashboard Cite

The field of acquired central nervous system neuroimmune demyelination in children is transforming. Recent advances in assay development, refinement of diagnostic criteria, increased biological insights provided by advanced neuroimaging techniques, and level 1A evidence for therapeutic efficacy of biological agents are re-defining diagnosis and care. Three distinct neuroimmune conditions-multiple sclerosis (MS), anti-myelin oligodendrocyte glycoprotein antibody associated disease (MOGAD) and anti-aquaporin-4 antibody neuromyelitis optica spectrum disorder (AQP4-NMOSD)-can now be distinguished, with human and animal model evidence supporting distinct pathobiological disease mechanisms. Development of highly effective therapies for adult-onset MS and NMOSD, with relapse rate suppression of greater than 90%, motivate advocacy for trials in children. However, clinical trials are challenged by the rarity of these conditions in the pediatric age group, necessitating new approaches to trial design including age-based trajectory modeling based on Phase 3 adult studies. Despite these limitations, the future for children and adolescents living with MS, MOGAD or NMOSD is far brighter than in years past, and will be brighter still if successful therapies to promote remyelination, enhance neuroprotection, and remediate cognitive deficits can be further accelerated.

show abstract

“…POMS is de ned as the manifestation of symptoms before/under the age of 16 or 18 [59]. According to our analysis, the frequency of FMS in POMS was higher than AOMS (Figure 5).…”

Section: Discussionmentioning

confidence: 76%

The global prevalence of familial multiple sclerosis: an updated systematic review and meta-analysis

Ehtesham

Rafie

Mosallaei

2021

Preprint

View full text Add to dashboard Cite

Background: Considering that familial multiple sclerosis (FMS) can reveal the extent to which genetic and environmental factors each involve in the etiopathogenesis of the disease, we performed an updated meta-analysis of the worldwide prevalence of FMS by addition of recent publications. Methods: A search in PubMed, Scopus, the ISI Web of Science, and Google Scholar up to 20 December 2020 was done. The inclusion criteria were based on the CoCoPop approach (condition, context, and population). The qualified studies entered the process of the meta-analysis by using comprehensive meta-analysis ver. 2 software.Results: The pooled prevalence of MS in relatives of 16179 FMS cases was estimated to be 11.8% (95% CI: 10.7-13) based on a random-effects model. The pooled mean age of disease onset in adult probands was calculated to be 28.7 years (95% CI: 27.2± 30.2). In 13 studies that reported the data of FMS in pediatrics (n=6636) and adults (n=877), the FMS prevalence was 10.8% (95% CI: 8.1-14.2) and 15.5% (95% CI: 13.8-17.4), respectively. Considering the data of 9 studies, the prevalence of FMS in males (n=5243) and females (n=11503) patients was calculated to be 13.7% (95% CI: 10.1-18.2) and 15.4% (95% CI: 10.3-22.4), respectively. The odds ratio of male/female in FMS cases was not statistically significant (OR= 0.9; 95% CI: 0.6-1.2, P=0.55). Subgroup analysis demonstrated a significant difference in the prevalence of FMS between the geographical areas (P= 0.007). The meta-regression model for FMS prevalence was significantly lower in terms of higher latitude (P< 0.001) and increased MS prevalence (P< 0.001). In contrast, meta-regression based on prevalence day was not statistically significant (P=0.29).Conclusions: The prevalence of FMS is more in the pediatric group than that of adults, is distinct between geographical areas, and diminishes with the increment of MS prevalence and latitude. Also, the symptoms initiate relatively at lower ages in FMS cases. By contrast with multifactorial diseases, our analysis unveiled that the prevalence of FMS was not more prevalent in men than women and the risk of MS development in relatives was not more when the affected proband was male.

show abstract

Epidemiology of pediatric multiple sclerosis: A systematic literature review and meta-analysis

Cited by 52 publications

References 46 publications

The global prevalence of familial multiple sclerosis: an updated systematic review and meta-analysis

The global prevalence of familial multiple sclerosis: an updated systematic review and meta-analysis

Paediatric multiple sclerosis and antibody-associated demyelination: clinical, imaging, and biological considerations for diagnosis and care

The global prevalence of familial multiple sclerosis: an updated systematic review and meta-analysis

Contact Info

Product

Resources

About