Background Perforin(PRF1)gene mutation can cause the onset of hemophagocytic lymphohistiocytosis(HLH).It has reported that PRFl Ala91 Val polymorphism was related with HLH risk.In the meta-analysis,we aim to evaluate ...Background Perforin(PRF1)gene mutation can cause the onset of hemophagocytic lymphohistiocytosis(HLH).It has reported that PRFl Ala91 Val polymorphism was related with HLH risk.In the meta-analysis,we aim to evaluate the association between PRF1 Ala91 Val polymorphism and HLH risk.Methods We accomplished a meta-analysis of six published case-control studies including 391 patients with HLH and 975 controls.We evaluated the quality of each study through Newcastle-Ottawa Scale(NOS).Data analysis was performed with Stata software.Results In general,all studies were of high quality(NOS score higher than 7).There were statistically significant between the PRFl Ala91 Val polymorphism and HLH risk though the pooled analysis[for Ala/Val vs.Ala/Ala:pooled odds ratio(OR)=3.22,95%confidence interval(Cl)1.08-9.56,P=0.035,random model;for Ala/Val+Val/Val vs.Ala/Ala:pooled OR=2.96,95%Cl 1.14-7.69,P=0.025,random model].Furthermore,sensitivity analysis also revealed a relationship between PRFl Ala91Val polymorphism and HLH risk(for Ala/Val vs.Ala/Ala:pooled OR=5.236,95%Cl 2.72-10.08,P<0.000,/2=12.1%,^heterogeneity=0-332;for Ala/Val+Val/Val vs.Ala/Ala,pooled OR=4.856,95%Cl 2.66-8.85,P<0.000,/2=5.9%,Pheterogeneity=0.373).Funnel plot and Egger’s test did not indicate obvious published bias(P=0.841 for Ala/Val vs.Ala/Ala;?=0.284 for Ala/Val+Val/Val vs.Ala/Ala).Conclusion This meta-analysis indicated that PRFl Ala91 Val polymorphism affects the factor for developing HLH and future studies of PRFl Ala91 Val on the onset of HLH will be guaranteed.展开更多
基金This study was funded by National Science and Technology Key Projects(No.2017ZX09304029001)the Beijing Municipal Science&Technology Commission(No.Z171100001017050)+4 种基金the National Natural Science Foundation of China(No.81700186,81800189)the Scientific Research Common Program of Beijing Municipal Commission of Education(No.KM201710025019)the Talent Training Project-Fostering Fund of National Natural Science Foundation of Beijing Children's Hospital,Capital Medical University(No.GPY201713)the Special Fund of The Pediatric Medical Coordinated Development Center of Beijing Hospitals Authority(No.XTZD20180202)the Guangdong Province Key Laboratory of Popular High Performance Computers of Shenzhen University(SZU-GDPHPCL2017).
文摘Background Perforin(PRF1)gene mutation can cause the onset of hemophagocytic lymphohistiocytosis(HLH).It has reported that PRFl Ala91 Val polymorphism was related with HLH risk.In the meta-analysis,we aim to evaluate the association between PRF1 Ala91 Val polymorphism and HLH risk.Methods We accomplished a meta-analysis of six published case-control studies including 391 patients with HLH and 975 controls.We evaluated the quality of each study through Newcastle-Ottawa Scale(NOS).Data analysis was performed with Stata software.Results In general,all studies were of high quality(NOS score higher than 7).There were statistically significant between the PRFl Ala91 Val polymorphism and HLH risk though the pooled analysis[for Ala/Val vs.Ala/Ala:pooled odds ratio(OR)=3.22,95%confidence interval(Cl)1.08-9.56,P=0.035,random model;for Ala/Val+Val/Val vs.Ala/Ala:pooled OR=2.96,95%Cl 1.14-7.69,P=0.025,random model].Furthermore,sensitivity analysis also revealed a relationship between PRFl Ala91Val polymorphism and HLH risk(for Ala/Val vs.Ala/Ala:pooled OR=5.236,95%Cl 2.72-10.08,P<0.000,/2=12.1%,^heterogeneity=0-332;for Ala/Val+Val/Val vs.Ala/Ala,pooled OR=4.856,95%Cl 2.66-8.85,P<0.000,/2=5.9%,Pheterogeneity=0.373).Funnel plot and Egger’s test did not indicate obvious published bias(P=0.841 for Ala/Val vs.Ala/Ala;?=0.284 for Ala/Val+Val/Val vs.Ala/Ala).Conclusion This meta-analysis indicated that PRFl Ala91 Val polymorphism affects the factor for developing HLH and future studies of PRFl Ala91 Val on the onset of HLH will be guaranteed.
