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Sci Rep ; 7(1): 17888, 2017 12 20.
Artigo em Inglês | MEDLINE | ID: mdl-29263366

RESUMO

Duchenne Muscular Dystrophy (DMD) is a severe muscle disorder caused by lack of dystrophin. Predictive biomarkers able to anticipate response to the therapeutic treatments aiming at dystrophin re-expression are lacking. The objective of this study is to investigate Matrix Metalloproteinase-9 (MMP-9) as predictive biomarker for Duchenne. Two natural history cohorts were studied including 168 longitudinal samples belonging to 66 patients. We further studied 1536 samples obtained from 3 independent clinical trials with drisapersen, an antisense oligonucleotide targeting exon 51: an open label study including 12 patients; a phase 3 randomized, double blind, placebo controlled study involving 186 patients; an open label extension study performed after the phase 3. Analysis of natural history cohorts showed elevated MMP-9 levels in patients and a significant increase over time in longitudinal samples. MMP-9 decreased in parallel to clinical stabilization in the 12 patients involved in the open label study. The phase 3 study and subsequent extension study clarified that the decrease in MMP-9 levels was not predictive of treatment response. These data do not support the inclusion of serum MMP-9 as predictive biomarker for DMD patients.


Assuntos
Biomarcadores/sangue , Metaloproteinase 9 da Matriz/sangue , Distrofia Muscular de Duchenne/sangue , Distrofia Muscular de Duchenne/genética , Oligonucleotídeos Antissenso/genética , Adolescente , Adulto , Criança , Pré-Escolar , Ensaios Clínicos Fase III como Assunto , Método Duplo-Cego , Distrofina/genética , Éxons/genética , Feminino , Humanos , Estudos Longitudinais , Masculino , Ensaios Clínicos Controlados Aleatórios como Assunto , Adulto Jovem
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