A meta-analysis of randomized double-blind clinical trials in CMT1A to assess the change from baseline in CMTNS and ONLS scales after one year of treatment
Identifieur interne : 003238 ( Main/Exploration ); précédent : 003237; suivant : 003239A meta-analysis of randomized double-blind clinical trials in CMT1A to assess the change from baseline in CMTNS and ONLS scales after one year of treatment
Auteurs : Jonas Mandel [France] ; Viviane Bertrand [France] ; Philippe Lehert [Australie, Belgique] ; Shahram Attarian [France] ; Laurent Magy [France] ; Joëlle Micallef [France] ; Ilya Chumakov [France] ; Catherine Scart-Grès [France] ; Mickael Guedj [France] ; Daniel Cohen [France]Source :
- Orphanet Journal of Rare Diseases [ 1750-1172 ] ; 2015.
Descripteurs français
- KwdFr :
- MESH :
- traitement médicamenteux : Maladie de Charcot-Marie-Tooth.
- usage thérapeutique : Acide ascorbique, Antioxydants.
- Essais contrôlés randomisés comme sujet, Humains.
English descriptors
- KwdEn :
- MESH :
- chemical , therapeutic use : Antioxidants, Ascorbic Acid.
- drug therapy : Charcot-Marie-Tooth Disease.
- Humans, Randomized Controlled Trials as Topic.
Abstract
CMT1A is the most common inherited peripheral neuropathy. There is currently no approved treatment. We performed a meta-analysis including four randomized, double-blind, Placebo-controlled clinical trials to assess the disease progression after one year under Placebo, Ascorbic Acid (AA) or PXT3003, a combination of three repurposed drugs. We observed a weak deterioration in patients under Placebo, well below the reported natural disease progression. Patients treated with AA were stable after one year but not significantly different from Placebo. Patients undergoing PXT3003 treatment showed an improvement in CMTNS and ONLS, statistically significant versus Placebo and potentially precursory of a meaningful change in the disease course.
Url:
DOI: 10.1186/s13023-015-0293-y
PubMed: 26070802
PubMed Central: 4482281
Affiliations:
- Australie, Belgique, France
- Limousin, Nouvelle-Aquitaine, Provence-Alpes-Côte d'Azur, Victoria (État)
- Limoges, Marseille, Melbourne
- Université de Melbourne
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Le document en format XML
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last="Lehert">Philippe Lehert</name><affiliation wicri:level="4"><nlm:aff id="Aff2">Faculty of Medicine, University of Melbourne, Melbourne, Australia</nlm:aff><country xml:lang="fr">Australie</country><wicri:regionArea>Faculty of Medicine, University of Melbourne, Melbourne</wicri:regionArea><placeName><settlement type="city">Melbourne</settlement><region type="état">Victoria (État)</region><settlement type="city">Melbourne</settlement></placeName><orgName type="university">Université de Melbourne</orgName></affiliation><affiliation wicri:level="1"><nlm:aff id="Aff3">Faculty of Economics, UCL Mons, Louvain, Belgium</nlm:aff><country xml:lang="fr">Belgique</country><wicri:regionArea>Faculty of Economics, UCL Mons, Louvain</wicri:regionArea><wicri:noRegion>Louvain</wicri:noRegion></affiliation></author><author><name sortKey="Attarian, Shahram" sort="Attarian, Shahram" uniqKey="Attarian S" first="Shahram" last="Attarian">Shahram Attarian</name><affiliation wicri:level="3"><nlm:aff id="Aff4">Centre de référence des maladies neuromusculaires et de la SLA, Pôle des neurosciences Cliniques, AP-HM et Aix Marseille Université, Marseille, France</nlm:aff><country xml:lang="fr">France</country><wicri:regionArea>Centre de référence des maladies neuromusculaires et de la SLA, Pôle des neurosciences Cliniques, AP-HM et Aix Marseille Université, Marseille</wicri:regionArea><placeName><region type="region">Provence-Alpes-Côte d'Azur</region><region type="old region">Provence-Alpes-Côte d'Azur</region><settlement type="city">Marseille</settlement></placeName></affiliation></author><author><name sortKey="Magy, Laurent" sort="Magy, Laurent" uniqKey="Magy L" first="Laurent" last="Magy">Laurent Magy</name><affiliation wicri:level="3"><nlm:aff id="Aff5">CHU de Limoges Hôpital Dupuytren, Limoges, France</nlm:aff><country xml:lang="fr">France</country><wicri:regionArea>CHU de Limoges Hôpital Dupuytren, Limoges</wicri:regionArea><placeName><region 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Chumakov</name><affiliation wicri:level="1"><nlm:aff id="Aff1">Pharnext SAS, Issy-les-Moulineaux, France</nlm:aff><country xml:lang="fr">France</country><wicri:regionArea>Pharnext SAS, Issy-les-Moulineaux</wicri:regionArea><wicri:noRegion>Issy-les-Moulineaux</wicri:noRegion><wicri:noRegion>Issy-les-Moulineaux</wicri:noRegion></affiliation></author><author><name sortKey="Scart Gres, Catherine" sort="Scart Gres, Catherine" uniqKey="Scart Gres C" first="Catherine" last="Scart-Grès">Catherine Scart-Grès</name><affiliation wicri:level="1"><nlm:aff id="Aff1">Pharnext SAS, Issy-les-Moulineaux, France</nlm:aff><country xml:lang="fr">France</country><wicri:regionArea>Pharnext SAS, Issy-les-Moulineaux</wicri:regionArea><wicri:noRegion>Issy-les-Moulineaux</wicri:noRegion><wicri:noRegion>Issy-les-Moulineaux</wicri:noRegion></affiliation></author><author><name sortKey="Guedj, Mickael" sort="Guedj, Mickael" uniqKey="Guedj M" first="Mickael" last="Guedj">Mickael Guedj</name><affiliation wicri:level="1"><nlm:aff id="Aff1">Pharnext SAS, Issy-les-Moulineaux, France</nlm:aff><country xml:lang="fr">France</country><wicri:regionArea>Pharnext SAS, Issy-les-Moulineaux</wicri:regionArea><wicri:noRegion>Issy-les-Moulineaux</wicri:noRegion><wicri:noRegion>Issy-les-Moulineaux</wicri:noRegion></affiliation></author><author><name sortKey="Cohen, Daniel" sort="Cohen, Daniel" uniqKey="Cohen D" first="Daniel" last="Cohen">Daniel Cohen</name><affiliation wicri:level="1"><nlm:aff id="Aff1">Pharnext SAS, Issy-les-Moulineaux, France</nlm:aff><country xml:lang="fr">France</country><wicri:regionArea>Pharnext SAS, Issy-les-Moulineaux</wicri:regionArea><wicri:noRegion>Issy-les-Moulineaux</wicri:noRegion><wicri:noRegion>Issy-les-Moulineaux</wicri:noRegion></affiliation></author></analytic><series><title level="j">Orphanet Journal of Rare Diseases</title><idno type="eISSN">1750-1172</idno><imprint><date when="2015">2015</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Antioxidants (therapeutic use)</term><term>Ascorbic Acid (therapeutic use)</term><term>Charcot-Marie-Tooth Disease (drug therapy)</term><term>Humans</term><term>Randomized Controlled Trials as Topic</term></keywords><keywords scheme="KwdFr" xml:lang="fr"><term>Acide ascorbique (usage thérapeutique)</term><term>Antioxydants (usage thérapeutique)</term><term>Essais contrôlés randomisés comme sujet</term><term>Humains</term><term>Maladie de Charcot-Marie-Tooth (traitement médicamenteux)</term></keywords><keywords scheme="MESH" type="chemical" qualifier="therapeutic use" xml:lang="en"><term>Antioxidants</term><term>Ascorbic Acid</term></keywords><keywords scheme="MESH" qualifier="drug therapy" xml:lang="en"><term>Charcot-Marie-Tooth Disease</term></keywords><keywords scheme="MESH" qualifier="traitement médicamenteux" xml:lang="fr"><term>Maladie de Charcot-Marie-Tooth</term></keywords><keywords scheme="MESH" qualifier="usage thérapeutique" xml:lang="fr"><term>Acide ascorbique</term><term>Antioxydants</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Humans</term><term>Randomized Controlled Trials as Topic</term></keywords><keywords scheme="MESH" xml:lang="fr"><term>Essais contrôlés randomisés comme sujet</term><term>Humains</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>CMT1A is the most common inherited peripheral neuropathy. There is currently no approved treatment. We performed a meta-analysis including four randomized, double-blind, Placebo-controlled clinical trials to assess the disease progression after one year under Placebo, Ascorbic Acid (AA) or PXT3003, a combination of three repurposed drugs. We observed a weak deterioration in patients under Placebo, well below the reported natural disease progression. Patients treated with AA were stable after one year but not significantly different from Placebo. Patients undergoing PXT3003 treatment showed an improvement in CMTNS and ONLS, statistically significant versus Placebo and potentially precursory of a meaningful change in the disease course.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Raeymaekers, P" uniqKey="Raeymaekers P">P Raeymaekers</name></author><author><name sortKey="Timmerman, V" uniqKey="Timmerman V">V Timmerman</name></author><author><name sortKey="Nelis, E" uniqKey="Nelis E">E Nelis</name></author><author><name sortKey="De Jonghe, P" uniqKey="De Jonghe P">P De Jonghe</name></author><author><name sortKey="Hoogendijk, Je" uniqKey="Hoogendijk J">JE Hoogendijk</name></author><author><name sortKey="Baas, F" uniqKey="Baas F">F Baas</name></author><author><name sortKey="Barker, Df" uniqKey="Barker D">DF Barker</name></author><author><name sortKey="Martin, Jj" uniqKey="Martin J">JJ Martin</name></author><author><name sortKey="De Visser, M" uniqKey="De Visser M">M De 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uniqKey="Mandel J" first="Jonas" last="Mandel">Jonas Mandel</name></noRegion><name sortKey="Attarian, Shahram" sort="Attarian, Shahram" uniqKey="Attarian S" first="Shahram" last="Attarian">Shahram Attarian</name><name sortKey="Bertrand, Viviane" sort="Bertrand, Viviane" uniqKey="Bertrand V" first="Viviane" last="Bertrand">Viviane Bertrand</name><name sortKey="Chumakov, Ilya" sort="Chumakov, Ilya" uniqKey="Chumakov I" first="Ilya" last="Chumakov">Ilya Chumakov</name><name sortKey="Cohen, Daniel" sort="Cohen, Daniel" uniqKey="Cohen D" first="Daniel" last="Cohen">Daniel Cohen</name><name sortKey="Guedj, Mickael" sort="Guedj, Mickael" uniqKey="Guedj M" first="Mickael" last="Guedj">Mickael Guedj</name><name sortKey="Magy, Laurent" sort="Magy, Laurent" uniqKey="Magy L" first="Laurent" last="Magy">Laurent Magy</name><name sortKey="Micallef, Joelle" sort="Micallef, Joelle" uniqKey="Micallef J" first="Joëlle" last="Micallef">Joëlle Micallef</name><name sortKey="Scart Gres, Catherine" sort="Scart Gres, Catherine" uniqKey="Scart Gres C" first="Catherine" last="Scart-Grès">Catherine Scart-Grès</name></country><country name="Australie"><region name="Victoria (État)"><name sortKey="Lehert, Philippe" sort="Lehert, Philippe" uniqKey="Lehert P" first="Philippe" last="Lehert">Philippe Lehert</name></region></country><country name="Belgique"><noRegion><name sortKey="Lehert, Philippe" sort="Lehert, Philippe" uniqKey="Lehert P" first="Philippe" last="Lehert">Philippe Lehert</name></noRegion></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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