Maximum inspiratory pressure as a clinically meaningful trial endpoint for neuromuscular diseases: a comprehensive review of the literature
Identifieur interne : 000013 ( France/Analysis ); précédent : 000012; suivant : 000014Maximum inspiratory pressure as a clinically meaningful trial endpoint for neuromuscular diseases: a comprehensive review of the literature
Auteurs : Benedikt Schoser [Allemagne] ; Edward Fong [États-Unis] ; Tarekegn Geberhiwot [Royaume-Uni] ; Derralynn Hughes [Royaume-Uni] ; John T. Kissel [États-Unis] ; Shyam C. Madathil ; David Orlikowski [France] ; Michael I. Polkey [Royaume-Uni] ; Mark Roberts [Royaume-Uni] ; Harm A. W. M. Tiddens [Pays-Bas] ; Peter Young [Allemagne]Source :
- Orphanet Journal of Rare Diseases [ 1750-1172 ] ; 2017.
Abstract
Respiratory muscle strength is a proven predictor of long-term outcome of neuromuscular disease (NMD), including amyotrophic lateral sclerosis, Duchenne muscular dystrophy, and spinal muscular atrophy. Maximal inspiratory pressure (MIP), a sensitive measure of respiratory muscle strength, one of several useful tests of respiratory muscle strength, is gaining interest as a therapeutic clinical trial endpoint for NMD. In this comprehensive review we investigate the use of MIP as a measure of respiratory muscle strength in clinical trials of therapeutics targeting respiratory muscle, examine the correlation of MIP with survival, quality of life, and other measures of pulmonary function, and outline the role of MIP as a clinically significantly meaningful outcome measure. Our analysis supports the utility of MIP for the early evaluation of respiratory muscle strength, especially of the diaphragm, in patients with NMD and as a surrogate endpoint in clinical trials of therapies for NMD.
Url:
DOI: 10.1186/s13023-017-0598-0
PubMed: 28302142
PubMed Central: 5353799
Affiliations:
- Allemagne, France, Pays-Bas, Royaume-Uni, États-Unis
- Angleterre, Bavière, District de Haute-Bavière, Midlands de l'Ouest, Ohio
- Birmingham, Munich
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PMC:5353799Le document en format XML
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W. M." last="Tiddens">Harm A. W. M. Tiddens</name><affiliation wicri:level="1"><nlm:aff id="Aff10"><institution-wrap><institution-id institution-id-type="GRID">grid.416135.4</institution-id><institution>Department of Pediatric Pulmonology and Allergology,</institution><institution>Sophia Children’s Hospital,</institution></institution-wrap>Rotterdam, The Netherlands</nlm:aff><country xml:lang="fr">Pays-Bas</country><wicri:regionArea>Rotterdam</wicri:regionArea></affiliation></author><author><name sortKey="Young, Peter" sort="Young, Peter" uniqKey="Young P" first="Peter" last="Young">Peter Young</name><affiliation wicri:level="1"><nlm:aff id="Aff11"><institution-wrap><institution-id institution-id-type="ISNI">0000 0001 2172 9288</institution-id><institution-id institution-id-type="GRID">grid.5949.1</institution-id><institution>Department of Sleep Medicine and Neuromuscular Disorders,</institution><institution>University of Münster,</institution></institution-wrap>Münster, Germany</nlm:aff><country xml:lang="fr">Allemagne</country><wicri:regionArea>Münster</wicri:regionArea></affiliation></author></analytic><series><title level="j">Orphanet Journal of Rare Diseases</title><idno type="eISSN">1750-1172</idno><imprint><date when="2017">2017</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Respiratory muscle strength is a proven predictor of long-term outcome of neuromuscular disease (NMD), including amyotrophic lateral sclerosis, Duchenne muscular dystrophy, and spinal muscular atrophy. Maximal inspiratory pressure (MIP), a sensitive measure of respiratory muscle strength, one of several useful tests of respiratory muscle strength, is gaining interest as a therapeutic clinical trial endpoint for NMD. In this comprehensive review we investigate the use of MIP as a measure of respiratory muscle strength in clinical trials of therapeutics targeting respiratory muscle, examine the correlation of MIP with survival, quality of life, and other measures of pulmonary function, and outline the role of MIP as a clinically significantly meaningful outcome measure. Our analysis supports the utility of MIP for the early evaluation of respiratory muscle strength, especially of the diaphragm, in patients with NMD and as a surrogate endpoint in clinical trials of therapies for NMD.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Perrin, C" uniqKey="Perrin C">C Perrin</name></author><author><name sortKey="Unterborn, Jn" uniqKey="Unterborn J">JN Unterborn</name></author><author><name sortKey="Ambrosio, Cd" uniqKey="Ambrosio C">CD Ambrosio</name></author><author><name sortKey="Hill, Ns" uniqKey="Hill N">NS Hill</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Epstein, Sk" uniqKey="Epstein S">SK Epstein</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Sansone, Va" uniqKey="Sansone V">VA Sansone</name></author><author><name sortKey="Gagnon, C" uniqKey="Gagnon C">C 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Wragg</name></author><author><name sortKey="Kyroussis, D" uniqKey="Kyroussis D">D Kyroussis</name></author><author><name sortKey="Aquilina, R" uniqKey="Aquilina R">R Aquilina</name></author><author><name sortKey="Moxham, J" uniqKey="Moxham J">J Moxham</name></author><author><name sortKey="Green, M" uniqKey="Green M">M Green</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Mcelvaney, G" uniqKey="Mcelvaney G">G McElvaney</name></author><author><name sortKey="Blackie, S" uniqKey="Blackie S">S Blackie</name></author><author><name sortKey="Morrison, Nj" uniqKey="Morrison N">NJ Morrison</name></author><author><name sortKey="Wilcox, Pg" uniqKey="Wilcox P">PG Wilcox</name></author><author><name sortKey="Fairbarn, Ms" uniqKey="Fairbarn M">MS Fairbarn</name></author><author><name sortKey="Pardy, Rl" uniqKey="Pardy R">RL Pardy</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Hart, N" uniqKey="Hart N">N Hart</name></author><author><name sortKey="Polkey, Mi" uniqKey="Polkey M">MI Polkey</name></author><author><name sortKey="Sharshar, T" uniqKey="Sharshar T">T Sharshar</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Morgan, Rk" uniqKey="Morgan R">RK Morgan</name></author><author><name sortKey="Mcnally, S" uniqKey="Mcnally S">S McNally</name></author><author><name sortKey="Alexander, M" uniqKey="Alexander M">M Alexander</name></author><author><name sortKey="Conroy, R" uniqKey="Conroy R">R Conroy</name></author><author><name sortKey="Hardiman, O" uniqKey="Hardiman O">O Hardiman</name></author><author><name sortKey="Costello, Rw" uniqKey="Costello R">RW Costello</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Khirani, S" uniqKey="Khirani S">S Khirani</name></author><author><name sortKey="Ramirez, A" uniqKey="Ramirez A">A Ramirez</name></author><author><name sortKey="Aubertin, G" uniqKey="Aubertin G">G Aubertin</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Fitting, Jw" uniqKey="Fitting J">JW Fitting</name></author><author><name sortKey="Paillex, R" uniqKey="Paillex R">R Paillex</name></author><author><name sortKey="Hirt, L" uniqKey="Hirt L">L Hirt</name></author><author><name sortKey="Aebischer, P" uniqKey="Aebischer P">P Aebischer</name></author><author><name sortKey="Schluep, M" uniqKey="Schluep M">M Schluep</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Stefanutti, D" uniqKey="Stefanutti D">D Stefanutti</name></author><author><name sortKey="Benoist, Mr" uniqKey="Benoist M">MR Benoist</name></author><author><name sortKey="Scheinmann, P" uniqKey="Scheinmann P">P Scheinmann</name></author><author><name sortKey="Chaussain, M" uniqKey="Chaussain M">M Chaussain</name></author><author><name sortKey="Fitting, Jw" uniqKey="Fitting J">JW Fitting</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Terzi, N" uniqKey="Terzi N">N Terzi</name></author><author><name sortKey="Orlikowski, D" uniqKey="Orlikowski D">D Orlikowski</name></author><author><name sortKey="Fermanian, C" uniqKey="Fermanian C">C Fermanian</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Gruis, Kl" uniqKey="Gruis K">KL Gruis</name></author><author><name sortKey="Lechtzin, N" uniqKey="Lechtzin N">N Lechtzin</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Syabbalo, N" uniqKey="Syabbalo N">N Syabbalo</name></author></analytic></biblStruct></listBibl></div1></back></TEI><affiliations><list><country><li>Allemagne</li><li>France</li><li>Pays-Bas</li><li>Royaume-Uni</li><li>États-Unis</li></country><region><li>Angleterre</li><li>Bavière</li><li>District de Haute-Bavière</li><li>Midlands de l'Ouest</li><li>Ohio</li></region><settlement><li>Birmingham</li><li>Munich</li></settlement></list><tree><noCountry><name sortKey="Madathil, Shyam C" sort="Madathil, Shyam C" uniqKey="Madathil S" first="Shyam C." last="Madathil">Shyam C. Madathil</name></noCountry><country name="Allemagne"><region name="Bavière"><name sortKey="Schoser, Benedikt" sort="Schoser, Benedikt" uniqKey="Schoser B" first="Benedikt" last="Schoser">Benedikt Schoser</name></region><name sortKey="Young, Peter" sort="Young, Peter" uniqKey="Young P" first="Peter" last="Young">Peter Young</name></country><country name="États-Unis"><noRegion><name sortKey="Fong, Edward" sort="Fong, Edward" uniqKey="Fong E" first="Edward" last="Fong">Edward Fong</name></noRegion><name sortKey="Kissel, John T" sort="Kissel, John T" uniqKey="Kissel J" first="John T." last="Kissel">John T. Kissel</name></country><country name="Royaume-Uni"><region name="Angleterre"><name sortKey="Geberhiwot, Tarekegn" sort="Geberhiwot, Tarekegn" uniqKey="Geberhiwot T" first="Tarekegn" last="Geberhiwot">Tarekegn Geberhiwot</name></region><name sortKey="Hughes, Derralynn" sort="Hughes, Derralynn" uniqKey="Hughes D" first="Derralynn" last="Hughes">Derralynn Hughes</name><name sortKey="Polkey, Michael I" sort="Polkey, Michael I" uniqKey="Polkey M" first="Michael I." last="Polkey">Michael I. Polkey</name><name sortKey="Roberts, Mark" sort="Roberts, Mark" uniqKey="Roberts M" first="Mark" last="Roberts">Mark Roberts</name></country><country name="France"><noRegion><name sortKey="Orlikowski, David" sort="Orlikowski, David" uniqKey="Orlikowski D" first="David" last="Orlikowski">David Orlikowski</name></noRegion></country><country name="Pays-Bas"><noRegion><name sortKey="Tiddens, Harm A W M" sort="Tiddens, Harm A W M" uniqKey="Tiddens H" first="Harm A. W. M." last="Tiddens">Harm A. W. M. Tiddens</name></noRegion></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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