Naeim Ehtesham1,2, Maryam Zare Rafie3, Meysam Mosallaei4. 1. Student Research Committee, University of Social Welfare and Rehabilitation Sciences, Koodakyar Alley, Daneshjoo Blvd., Evin St, Tehran, Iran. na.ehtesham@uswr.ac.ir. 2. Department of Genetics and Molecular Biology, School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran. na.ehtesham@uswr.ac.ir. 3. School of Medicine, Zanjan University of Medical Sciences, Zanjan, Iran. 4. Department of Genetics and Molecular Biology, School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran.
Abstract
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.
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.
Authors: Zahra Salehi; Amir Almasi-Hashiani; Mohammad Ali Sahraian; Sharareh Eskandarieh Journal: Mult Scler Relat Disord Date: 2020-05-14 Impact factor: 4.339
Authors: Loukas Moutsianas; Luke Jostins; Ashley H Beecham; Alexander T Dilthey; Dionysia K Xifara; Maria Ban; Tejas S Shah; Nikolaos A Patsopoulos; Lars Alfredsson; Carl A Anderson; Katherine E Attfield; Sergio E Baranzini; Jeffrey Barrett; Thomas M C Binder; David Booth; Dorothea Buck; Elisabeth G Celius; Chris Cotsapas; Sandra D'Alfonso; Calliope A Dendrou; Peter Donnelly; Bénédicte Dubois; Bertrand Fontaine; Lars Fugger; An Goris; Pierre-Antoine Gourraud; Christiane Graetz; Bernhard Hemmer; Jan Hillert; Ingrid Kockum; Stephen Leslie; Christina M Lill; Filippo Martinelli-Boneschi; Jorge R Oksenberg; Tomas Olsson; Annette Oturai; Janna Saarela; Helle Bach Søndergaard; Anne Spurkland; Bruce Taylor; Juliane Winkelmann; Frauke Zipp; Jonathan L Haines; Margaret A Pericak-Vance; Chris C A Spencer; Graeme Stewart; David A Hafler; Adrian J Ivinson; Hanne F Harbo; Stephen L Hauser; Philip L De Jager; Alastair Compston; Jacob L McCauley; Stephen Sawcer; Gil McVean Journal: Nat Genet Date: 2015-09-07 Impact factor: 38.330