Guidelines for the preclinical in vivo evaluation of pharmacological active drugs for ALS MND: Report on the 142nd ENMC international workshop
A transgenic animal model for anterior horn cell loss was established in 1994. This model is based on the insertion of a high copy number of disease-causing human Cu Zn SOD mutations into the intact mouse genome. It serves to establish hypotheses for the pathogenesis of anterior horn cell death, but...
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Published in | Amyotrophic lateral sclerosis Vol. 8; no. 4; pp. 217 - 223 |
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Main Authors | , , , , , , , , , , , , , |
Format | Journal Article |
Language | English |
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England
Informa UK Ltd
2007
Taylor & Francis |
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Abstract | A transgenic animal model for anterior horn cell loss was established in 1994. This model is based on the insertion of a high copy number of disease-causing human Cu Zn SOD mutations into the intact mouse genome. It serves to establish hypotheses for the pathogenesis of anterior horn cell death, but also to test potential pharmacological approaches to therapy in human ALS. Today, more than 100 - published and unpublished - compounds have been tested in this animal model, a large part of them being reported as successful. However, it proved to be difficult to translate these therapeutic successes in the animal model into human trials. Also, a number of disease-modifying strategies were difficult to reproduce, even by the same group. On the other hand, the step from mice to men means a huge investment for the sponsors of clinical trials and the scientific community. Therefore, establishment of standard methods for drug testing in ALS models is mandatory. In this workshop, clinical and preclinical researchers established in the field of ALS MND met in Holland in March 2006 in order to establish guidelines for the community for drug testing in mouse models. |
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AbstractList | A transgenic animal model for anterior horn cell loss was established in 1994. This model is based on the insertion of a high copy number of disease-causing human Cu/Zn SOD mutations into the intact mouse genome. It serves to establish hypotheses for the pathogenesis of anterior horn cell death, but also to test potential pharmacological approaches to therapy in human ALS. Today, more than 100 - published and unpublished - compounds have been tested in this animal model, a large part of them being reported as successful. However, it proved to be difficult to translate these therapeutic successes in the animal model into human trials. Also, a number of disease-modifying strategies were difficult to reproduce, even by the same group. On the other hand, the step from mice to men means a huge investment for the sponsors of clinical trials and the scientific community. Therefore, establishment of standard methods for drug testing in ALS models is mandatory. In this workshop, clinical and preclinical researchers established in the field of ALS/MND met in Holland in March 2006 in order to establish guidelines for the community for drug testing in mouse models. |
Author | Scott, Sean ENMC GROUP FOR THE ESTABLISHMENT OF GUIDELINES FOR THE CONDUCT OF PRECLINICAL AND PROOF OF CONCEPT STUDIES IN ALS MND MODELS Van Den Berg, Leonard H. Ludolph, Albert C. Meininger, Vincent Robberecht, Wim Bendotti, Caterina Meyer, Thomas Blaugrund, Eran Martin, Joanne Moussaoui, Saliha Hengerer, Bastian Löffler, Jean-Philippe Silani, Vincenzo |
Author_xml | – sequence: 1 givenname: Albert C. surname: Ludolph fullname: Ludolph, Albert C. email: albert.ludolph@medizin.uni-ulm.de organization: 1Department of Neurology, University of Ulm, Ulm, Germeny – sequence: 2 givenname: Caterina surname: Bendotti fullname: Bendotti, Caterina email: albert.ludolph@medizin.uni-ulm.de organization: 1Department of Neurology, University of Ulm, Ulm, Germeny – sequence: 3 givenname: Eran surname: Blaugrund fullname: Blaugrund, Eran email: albert.ludolph@medizin.uni-ulm.de organization: 1Department of Neurology, University of Ulm, Ulm, Germeny – sequence: 4 givenname: Bastian surname: Hengerer fullname: Hengerer, Bastian email: albert.ludolph@medizin.uni-ulm.de organization: 1Department of Neurology, University of Ulm, Ulm, Germeny – sequence: 5 givenname: Jean-Philippe surname: Löffler fullname: Löffler, Jean-Philippe email: albert.ludolph@medizin.uni-ulm.de organization: 1Department of Neurology, University of Ulm, Ulm, Germeny – sequence: 6 givenname: Joanne surname: Martin fullname: Martin, Joanne email: albert.ludolph@medizin.uni-ulm.de organization: 1Department of Neurology, University of Ulm, Ulm, Germeny – sequence: 7 givenname: Vincent surname: Meininger fullname: Meininger, Vincent email: albert.ludolph@medizin.uni-ulm.de organization: 1Department of Neurology, University of Ulm, Ulm, Germeny – sequence: 8 givenname: Thomas surname: Meyer fullname: Meyer, Thomas email: albert.ludolph@medizin.uni-ulm.de organization: 1Department of Neurology, University of Ulm, Ulm, Germeny – sequence: 9 givenname: Saliha surname: Moussaoui fullname: Moussaoui, Saliha email: albert.ludolph@medizin.uni-ulm.de organization: 1Department of Neurology, University of Ulm, Ulm, Germeny – sequence: 10 givenname: Wim surname: Robberecht fullname: Robberecht, Wim email: albert.ludolph@medizin.uni-ulm.de organization: 1Department of Neurology, University of Ulm, Ulm, Germeny – sequence: 11 givenname: Sean surname: Scott fullname: Scott, Sean email: albert.ludolph@medizin.uni-ulm.de organization: 1Department of Neurology, University of Ulm, Ulm, Germeny – sequence: 12 givenname: Vincenzo surname: Silani fullname: Silani, Vincenzo email: albert.ludolph@medizin.uni-ulm.de organization: 1Department of Neurology, University of Ulm, Ulm, Germeny – sequence: 13 givenname: Leonard H. surname: Van Den Berg fullname: Van Den Berg, Leonard H. email: albert.ludolph@medizin.uni-ulm.de organization: 1Department of Neurology, University of Ulm, Ulm, Germeny – sequence: 14 surname: ENMC GROUP FOR THE ESTABLISHMENT OF GUIDELINES FOR THE CONDUCT OF PRECLINICAL AND PROOF OF CONCEPT STUDIES IN ALS MND MODELS fullname: ENMC GROUP FOR THE ESTABLISHMENT OF GUIDELINES FOR THE CONDUCT OF PRECLINICAL AND PROOF OF CONCEPT STUDIES IN ALS MND MODELS email: albert.ludolph@medizin.uni-ulm.de organization: 1Department of Neurology, University of Ulm, Ulm, Germeny |
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SubjectTerms | Amyotrophic Lateral Sclerosis - drug therapy Animals Disease Models, Animal Drug Evaluation, Preclinical - methods Humans Mice Mice, Transgenic Superoxide Dismutase - genetics |
Title | Guidelines for the preclinical in vivo evaluation of pharmacological active drugs for ALS MND: Report on the 142nd ENMC international workshop |
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