Evaluating enzyme replacement therapies for Anderson-Fabry disease : commentary on a recent report

Anderson-Fabry disease (AFD) is a rare lysosomal storage disorder. Randomized controlled clinical trials (RCTs) are preferred as the highest category of evidence, but limited availability of robust evidence in rare diseases may necessitate the use of less rigorous evidence. An analysis of cohort stu...

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Detalles Bibliográficos
Autores: Giugliani, Roberto, Westwood, Stephanie, Wellhoefer, Hartmann, Schenk, Jörn Magnus, Gurevich, Andrey, Kampmann, Christoph
Tipo de recurso: artículo
Estado:Versión publicada
Fecha de publicación:2018
País:Brasil
Institución:Universidade Federal do Rio Grande do Sul (UFRGS)
Repositorio:Repositório Institucional da UFRGS
Idioma:inglés
OAI Identifier:oai:www.lume.ufrgs.br:10183/194775
Acceso en línea:http://hdl.handle.net/10183/194775
Access Level:acceso abierto
Palabra clave:Terapia de reposição de enzimas
Doença de Fabry
Agalsidase alfa
Agalsidase beta
Anderson-Fabry disease
Enzyme replacement therapy
Descripción
Sumario:Anderson-Fabry disease (AFD) is a rare lysosomal storage disorder. Randomized controlled clinical trials (RCTs) are preferred as the highest category of evidence, but limited availability of robust evidence in rare diseases may necessitate the use of less rigorous evidence. An analysis of cohort studies of enzyme replacement therapies for AFD published in 2017 by El Dib and coworkers made treatment recommendations that contradict previously published findings from RCTs and a systematic Cochrane review. Our commentary outlines concerns regarding selection criteria and statistical methods with their analysis.