Enzyme replacement therapy for Anderson-Fabry disease: A complementary overview of a Cochrane publication through a linear regression and a pooled analysis of proportions from cohort studies
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{"title"=>"Enzyme replacement therapy for Anderson-Fabry disease: A complementary overview of a Cochrane publication through a linear regression and a pooled analysis of proportions from cohort studies", "type"=>"journal", "authors"=>[{"first_name"=>"Regina", "last_name"=>"El Dib", "scopus_author_id"=>"16039445900"}, {"first_name"=>"Huda", "last_name"=>"Gomaa", "scopus_author_id"=>"57190393483"}, {"first_name"=>"Alberto", "last_name"=>"Ortiz", "scopus_author_id"=>"57200124502"}, {"first_name"=>"Juan", "last_name"=>"Politei", "scopus_author_id"=>"6507209754"}, {"first_name"=>"Anil", "last_name"=>"Kapoor", "scopus_author_id"=>"56844643500"}, {"first_name"=>"Fellype", "last_name"=>"Barreto", "scopus_author_id"=>"13605277200"}], "year"=>2017, "source"=>"PLoS ONE", "identifiers"=>{"pmid"=>"28296917", "doi"=>"10.1371/journal.pone.0173358", "sgr"=>"85015412087", "isbn"=>"1111111111", "scopus"=>"2-s2.0-85015412087", "issn"=>"19326203", "pui"=>"614792212"}, "id"=>"7ce2bb13-7064-3643-a886-b0daf53397d8", "abstract"=>"Background Anderson-Fabrydisease(AFD)isanX-linkedrecessiveinbornerrorof glycosphingolipid metabolismcausedbyadeficiencyofalpha-galactosidaseA.Renalfailure,heartand cere-brovascularinvolvementreducesurvival.ACochranereviewprovidedlittleevidenceon the useofenzymereplacementtherapy(ERT).Wenowcomplementthisreviewthrougha lin-earregressionandapooledanalysisofproportionsfromcohort studies. Objectives ToevaluatetheefficacyandsafetyofERTfor AFD. Materialsand methods Forthesystematicreview,aliteraturesearchwasperformed,frominceptiontoMarch 2016, usingMedline,EMBASEandLILACS.Inclusioncriteriawerecohortstudies,patients with AFDonERTornaturalhistory,andatleastonepatient-importantoutcome(all-cause mor-tality,renal,cardiovascularorcerebrovascularevents,andadverseevents)reported. The pooledproportionandtheconfidenceinterval(CI)areshownforeachoutcome.Simple lin-earregressionsforcompositeendpointswere performed. Results 77cohortstudiesinvolving15,305participantsprovedeligible.Thepooledproportions were asfollows:a)forrenalcomplications,agalsidasealfa15.3%[95%CI0.048,0.303;I2 = 77.2%,p=0.0005];agalsidasebeta6%[95%CI0.04,0.07;I2=notapplicable]; and untreatedpatients21.4%[95%CI0.1522,0.2835;I2=89.6%,p<0.0001].Effect differences favoredagalsidasebetacomparedtountreatedpatients;b)forcardiovascular complica-tions,agalsidasealfa28%[95%CI0.07,0.55;I2=96.7%,p<0.0001];agalsidasebeta 7% [95%CI0.05,0.08;I2=notapplicable];anduntreatedpatients26.2%[95%CI0.149, 0.394; I2=98.8%,p<0.0001].Effectdifferencesfavoredagalsidasebetacomparedto untreated patients;andc)forcerebrovascularcomplications,agalsidasealfa11.1%[95%CI 0.058, 0.179;I2=70.5%,p=0.0024];agalsidasebeta3.5%[95%CI0.024,0.046;I2=0%,p = 0.4209];anduntreatedpatients18.3%[95%CI0.129,0.245;I2=95%p<0.0001]. Effect differencesfavoredagalsidasebetaoveragalsidasealfaoruntreatedpatients.A linear regressionshowedthatFabrypatientsreceivingagalsidasealfaaremorelikelyto have higherratesofcompositeendpointscomparedtothosereceivingagalsidase beta. Conclusions Agalsidasebetaisassociatedtoasignificantlylowerincidenceofrenal,cardiovascular and cerebrovasculareventsthannoERT,andtoasignificantlylowerincidenceof cerebrovascu-lareventsthanagalsidasealfa.Inviewoftheseresults,theuseofagalsidasebetafor preventing major", "link"=>"http://www.mendeley.com/research/enzyme-replacement-therapy-andersonfabry-disease-complementary-overview-cochrane-publication-through", "reader_count"=>26, "reader_count_by_academic_status"=>{"Unspecified"=>1, "Professor > Associate Professor"=>2, "Librarian"=>1, "Researcher"=>6, "Student > Ph. D. Student"=>1, "Student > Postgraduate"=>1, "Student > Master"=>5, "Other"=>2, "Student > Bachelor"=>5, "Professor"=>2}, "reader_count_by_user_role"=>{"Unspecified"=>1, "Professor > Associate Professor"=>2, "Librarian"=>1, "Researcher"=>6, "Student > Ph. D. Student"=>1, "Student > Postgraduate"=>1, "Student > Master"=>5, "Other"=>2, "Student > Bachelor"=>5, "Professor"=>2}, "reader_count_by_subject_area"=>{"Unspecified"=>1, "Engineering"=>1, "Biochemistry, Genetics and Molecular Biology"=>5, "Nursing and Health Professions"=>1, "Medicine and Dentistry"=>6, "Agricultural and Biological Sciences"=>3, "Pharmacology, Toxicology and Pharmaceutical Science"=>4, "Computer Science"=>2, "Immunology and Microbiology"=>1, "Economics, Econometrics and Finance"=>2}, "reader_count_by_subdiscipline"=>{"Engineering"=>{"Engineering"=>1}, "Medicine and Dentistry"=>{"Medicine and Dentistry"=>6}, "Immunology and Microbiology"=>{"Immunology and Microbiology"=>1}, "Economics, Econometrics and Finance"=>{"Economics, Econometrics and Finance"=>2}, "Agricultural and Biological Sciences"=>{"Agricultural and Biological Sciences"=>3}, "Computer Science"=>{"Computer Science"=>2}, "Nursing and Health Professions"=>{"Nursing and Health Professions"=>1}, "Biochemistry, Genetics and Molecular Biology"=>{"Biochemistry, Genetics and Molecular Biology"=>5}, "Unspecified"=>{"Unspecified"=>1}, "Pharmacology, Toxicology and Pharmaceutical Science"=>{"Pharmacology, Toxicology and Pharmaceutical Science"=>4}}, "reader_count_by_country"=>{"Brazil"=>1, "Costa Rica"=>1}, "group_count"=>4}

Scopus | Further Information

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