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112 results on '"H. Staunton"'

1. SMA - TREATMENT

Author

A. Nascimento, J. Day, N. Deconinck, E. Mazzone, M. Oskoui, K. Saito, C. Vuillerot, G. Baranello, O. Boespflug-Tanguy, N. Goemans, J. Kirschner, A. Kostera-Pruszczyk, L. Servais, M. Gerber, K. Gorni, C. Martin, R. Scalco, H. Staunton, W. Yeung, and E. Mercuri

Subjects
Neurology, Pediatrics, Perinatology and Child Health, Neurology (clinical), Genetics (clinical)
Published
2021
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2. SMA – THERAPY

Author

J. Day, G. Baranello, O. Boespflug-Tanguy, S. Borell, N. Goemans, J. Kirschner, R. Masson, M. Pera, L. Servais, S. Fuhrer, M. Gerber, K. Gorni, H. Kletzl, C. Martin, R. Scalco, H. Staunton, W. Yeung, and E. Mercuri

Subjects
Neurology, Pediatrics, Perinatology and Child Health, Neurology (clinical), Genetics (clinical)
Published
2020
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3. P.196Estimating clinically meaningful change thresholds in the NORTH STAR ambolatory assessment (NSAA) and four-stair climb (4SC) in Duchenne muscular dystrophy (DMD)

Author

cTAP, Zhiwen Yao, M. Rabbia, R. Ong, Brenda Wong, I. Dieye, H. Staunton, James Signorovitch, Gautam Sajeev, and S. Ward

Subjects
medicine.medical_specialty, Physical medicine and rehabilitation, Neurology, business.industry, Duchenne muscular dystrophy, Pediatrics, Perinatology and Child Health, medicine, Climb, Neurology (clinical), medicine.disease, business, Genetics (clinical)
Published
2019
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4. HaemoPREF: Further evaluation of patient perception and preference for treatment in a real world setting

Author

E. Brohan, Ana C Gordo, Michael Cicchetti, Dean Spurden, Joseph C. Cappelleri, H. Staunton, Santiago Bonanad, Martin Schulz, and Chloe Tolley

Subjects
Adult, Male, medicine.medical_specialty, Multivariate statistics, Haemophilia, Patients, Psychometrics, Intraclass correlation, 030204 cardiovascular system & hematology, Hemophilia A, Spearman's rank correlation coefficient, Preference, Correlation, 03 medical and health sciences, 0302 clinical medicine, Cronbach's alpha, Patient-Reported Outcome, HaemoPREF, Germany, Surveys and Questionnaires, medicine, Humans, Genetics (clinical), Aged, Factor VIII, business.industry, Ease of Use, Reproducibility of Results, Patient Preference, Hematology, General Medicine, Middle Aged, medicine.disease, Treatment Adherence and Compliance, Cross-Sectional Studies, Convergent validity, Italy, Patient Satisfaction, Spain, Physical therapy, Treatment Satisfaction, Patient-reported outcome, Perception, business, 030215 immunology
Abstract

IntroductionAdherence to haemophilia A (HA) treatment may be influenced by patients' beliefs about their condition and treatment. Furthermore, difficulties administering treatment may lead to poor adherence. New treatment strategies aim to reduce the burden associated with administration and to improve patient perception of treatment, which, in turn, increase adherence levels. AimsThis study aimed to examine patient perception of HA treatment and related factors using patient-reported outcome (PRO) questionnaires and to confirm the psychometric properties of a recently developed questionnaire, the HaemoPREF. MethodsA non-interventional, cross-sectional, questionnaire study was conducted with adult HA patients in Spain (n=31), Germany (n=10) and Italy (n=48), who were using ReFacto AF with the FuseNGo administration device. Patients completed the HaemoPREF and other questionnaires measuring related constructs: treatment adherence, satisfaction and well-being, online at two time points. Correlational, regression and psychometric analyses were conducted. ResultsPRO scores indicated that patients are satisfied with and adherent to their treatment. Multivariate regression of the HaemoPREF global score identified a number of significant predictors (P.05). The HaemoPREF Global Score had a moderate relationship with subscales on the related questionnaires (mean correlation=0.43; range=0.39-0.48). The HaemoPREF demonstrated good test-retest reliability (intraclass correlation coefficient=0.82), internal consistency reliability (Cronbach's alpha range=0.69-0.82) and convergent validity with measures of treatment satisfaction (Spearman correlation coefficient, r=.48) and well-being (r=.41). ConclusionThe findings suggest that patients using ReFacto AF with FuseNGo were satisfied with and adherent to their treatment. The HaemoPREF can identify important concepts relating to patient treatment experience in HA.

Published
2017

5. Efficiency testing of motors powered from pulse-width modulated adjustable speed drives

Author

H. Staunton, D.A. Casada, J.D. Kueck, and M.C. Webb

Subjects
Engineering, business.industry, Energy Engineering and Power Technology, Volt, Horsepower, Data acquisition, Direct torque control, Control theory, Torque, Electrical and Electronic Engineering, business, Pulse width modulated, Pulse-width modulation, Induction motor
Abstract

This is a report of efficiency testing of an induction motor powered from three different pulse width modulated adjustable speed drives. The motor was operated at a range of speeds and torques, and each drive was operated at the highest, lowest, and an in-between carrier frequency. The motor was a typical NEMA design B motor, and the drives were typical, industrial scalar drives using the default volts/Hz setting. The testing showed that the drive efficiencies remained above 90% until torque was lowered to below 20% of rated torque. Combined motor and drive efficiencies remained above 80% until speeds or horsepower loads were lowered to below 20% of rated torque. The test was performed using a unique data acquisition scheme that permitted acquisition of a large number of torque settings for each speed selection.

Published
2000
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6. Fourth meeting of the European Neurological Society 25–29 June 1994 Barcelona, Spain

Author

H. Hattig, C. Delli Pizzi, M. C. Addonizio, Michelle Davis, A. R. Giovagnoli, L. Florensa, M. Roth, J. de Kruijk, Francisco Lacruz, Ph. Dewailly, A. Toygar, C. Avendano, P.P. De Deyn, J. F. Hurtevent, F. Lomeila, T. W. Wong, Gordon T. Plant, M. Bud, H. J. Willison, DH Miller, D. W. Langdon, R. Cioni, J. Servan, A. Kaygisiz, E. Racadot, D. B. Schens, E. Picciola, L. Falip, C. Bouchard, J. Jotova, A. Jorge-Santamaria, P. Misra, A. Dufour, C. P. Panagopoulos, A. Venneri, B. Sredni, B. Angelard, M. Janelidze, M. Carreno, J. Obenberger, J. Pouget, H. W. Moser, R. Kaufmann, J. A. Molina, D. Linden, A. Martin Urda, E. Uvestad, A. Krone, J. P. Cochin, J. Mallecourt, A. Cambon-Thomsen, K. Violleau, P. Osschmann, A. M. Durocher, E. Bussaglia, D. M. Danielle, H. Efendi, C. Van Broeckhoven, K. G. Jordan, W. Rautenberg, C. Iniguez, J. M. Delgado, Graham Watson, M. Lawden, Gareth J. Barker, K. Stiasny, James T. Becker, G. Campanella, E. Peghi, A. Poli, A. Haddad, T. Yamawaki, Giacomo P. Comi, S. Sotgiu, B. Ersmark, A. Pomes, M. Ziegler, P. Ferrante, P. Ruppi, H. KuÇukoglu, R. Bouton, U. K. Rinne, P. Vieregge, M. Dary, P. Giunti, Peter J. Goadsby, S. Jung, E. Secor, A. Steinberg, N. Vila, M. A. Hernandez, M. Cursi, A. Enqelhardt, A. Engelhardt, J. Veitch, F. Di Silverio, F. Arnaud, B. Neundörfer, R. Brucher, Dominique Caparros-Lefebvre, B. Meyer, Marianne Dieterich, M. H. Snidaro, R. Gomez, R. Cerbo, M. Ragno, J. M. Vance, S. Nemni, A. Caliskan, F. Barros, I. Velcheva, D. Ceballos-Baumann, V. Barak, A. Avila, N. Antonova, F. Resche, S. Pappata, L. Varela, S. R. Silveira Santos, A. Cammarota, L. Naccache, Y. Nara, E. Tournier-Lasserves, R. Mobner, T. Chase, A. Ensenyat, J. Ulrich, G. Giegerich, M. Rother, M. Revilla, N. Nitschke, K. Honczarenko, E. Basart Tarrats, J. Blin, B. Jacob, J. Santamaria, S. Knezevic, J. L. Castillo, M. Antem, J. Colomer, O. Busse, Didier Hannequin, S. Carrier, J. B. Ruidavets, C. Rozman, J. Bogoussslavsky, J. Pascual Calvet, E. Monros, J. M. Polo, M. Zucconl, Javier Muruzabal, R. R. Allen, R. Rivolta, K. Haugaard, A. Nespolo, K. Hoang-Xuang, G. Bussone, T. Avramidis, E. Corsini, Christiana Franke, T. Vinogradova, H. Boot, K. Vestergaard, G. H. Jansen, N. Argentino, M. Raltzig, W. Linssen, Mark B. Pepys, P. Roblot, L. Lauritzen, E. Fainardi, D. Morin, T. X. Arbizu Urdiain, J. Wollenhaupt, S. Bostantjopoulou, G. Pavesi, A. D. Forman, Giovanni Fabbrini, D. Jean, J. J. Archelos, M. I. Blanchs, M. Del Gobbo, Anna Carla Turconi, Ch. Derouesné, Elio Scarpini, A. Visbeck, P. Castejon, J. P. Renou, F. Mounier-Vehier, G. Potagas, Ch. Duyckaerts, A. Filla, R. Schneider, G. Ronen, K. Nagata, J. P. Vedel, A. Henneberg, G. van Melle, C. Baratti, H. Knott, M. C. Prevett, A. Bes, B. Metin, Jos V. Reempts, L. Martorell, Mefkure Eraksoy, H. O. Handwerker, D. S. Younger, O. Oktem, D. Frongillo, C. Soriano-Soriano, L. Niehaus, F. Zipp, A. Tartaro, S Newman, R. H. Browne, P. Davous, R. Sanchez, M. Muros, M. E. Kornhuber, A. Lavarone, M. Mohr, M. R. Garcia, S. Russell, H. Kellar-Wood, M. R. Tola, B. Ostermeyer, Ch. Tzekov, K. Sartor, E. B. Ringelstein, P. P. Gazzaniga, Paul Krack, H. Fidaner, H. Rico, T. Dbaiss, F. Alameda, E. Torchiana, L. Rumbach, I. Charques, J. M. Bogaard, C. D. Frith, L. J. Rappelle, R. Brenner, A. Joutel, K. Fuxe, G. HÄcker, M. J. Blaser, J. Valls-SolÇ, G. Ulm, M. Alberdi, A. Bock, F. W. Bertelsmann, U. Wieshmann, J. Visa, J. R. Lupski, D. D'Amico, L. M. P. Ramos, A. A. Vanderbark, R. Horn, M. Warmuth, Dietmar Kühne, Mark S. Palmer, C. Ehrenheim, E. Canga, S. Viola, O. Scarpino, P. Naldi, R. Almeida, A. A. Raymond, J. Gamez, Stephan Arnold, A. DiGiovanni, J. Dalmau, C. C. Chari, H. F. Beer, J. C. Koetsier, J. Iriarte, E. Yunis, J. Casadevall, E. Le Guern, E. Stenager, S. R. Benbadis, J. M. Warter, F. Burklin, I. Theodorou, L. Johannesen, G. A. Graveland, X. Leclerc, I. Vecchio, L. Ozelius, G. Nicoletti, R. K. Gherardi, E. Esperet, M. L. Delodovici, F. Cattin, F. Paiau, Giorgio Sacilotto, C. A. J. Broere, D. Chavdarov, J. P. Willmer, C. H. Hawkes, Th. Naegele, E. Ellie, E. Dartigues, M. J. Guardiola, S. Hesse, Z. Levic, Marco Rovaris, P. Saugeir-Veber, B. A. Yaqub, H. F. Durwen, R. Larumbe, J. Ballabrina, M. Sendtner, J. Röther, M. Horstink, C. Kluglein, M.P. Montesi, H. Apaydin, J. Montoya, E. Waubant, Ch. Verellen-Dunoulin, A. Nicolai, J. Lopez-Delval, R. Lemon, G. Cantinho, E. Granieri, A. Zeviani, Wolfgang H. Oertel, U. Ficola, V. Di Piero, V. Fragola, K. Sabev, M. V. Guitera, I. Turki, F. Bolgert, P. Ingrand, J. M. Gobernado, L. M. E. Grimaldi, S. Baybas, B. Eymard, Y. Rolland, Y. Robitaille, Ta. Pampols, P. J. Koehler, A. Carroacedo, J. Vilchez, S. Di Vittorio, I. R. Rise, T. Nagy, M. Kuffner, E. Palazzini, A. Ott, J. Pruim, T. X. Arbizu, E. Manetti, C. Cervera, S. Felber, G. Gursoy, J. Scholz, G. A. Buscaino, M. S. Chen, A. Pascual, J. Hazan, J. U. Gajda, J. G. Cea, G. Bottini, G. Damalik, F. Le Doze, G. Bonaldi, J. M. Hew, C. Messina, A. M. Kennedy, J. M. Carney, N. M. F. Murray, M. Parent, M. Koepp, V. Dimova, D. De Leo, K. Jellinger, G. Salemi, S. Mientus, M. L. Hansen, F. Mazzucchelli, J. Vieth, M. Mauri, E. Bartels, L. Johannsen, C. Humphreys, J. Emile, D. N. Landon, E. Kansu, R. Sanchez-Pernaute, Rsj Frackowiak, M. Gonzalez Torres, L. Oller, C. Machedo, J. Kother, M. Billiard, H. Durak, T. Schindler, A. Frank, A. Uncini, A. Sbriccoli, C. Farinas, D. W. Paty, N. Fast, A. T. Zangaladze, A. Kerkhofs, J. M. Pino Garcia, I. De la Fuente, B. Marini, L. Gomez, I. Rubio, Alessandra Bardoni, C. Brodie, P. Acin, U. Sliwka, S. A. Hawkins, S. Tardieu, F. Vitullo, J. M. Pereira Monteino, R. Gagliardi, T. Jezewski, A. Cano, T. Lempert, F. Abad Alegria, G. Rotondo, D. Ince, C. Martinez Parra, Y. Huang, H. Luders, Y. Steinvil, F. G. A. Van Der Meche, R. Bianchi, A. Sanchez, T. Sevilla, J. M. Ketelslegers, A. Domzal-Stryga, M. Pandolfo, M. O. Josse, K. W. Neff, I. Blanco, G. W. Bruyn, O. W. Witte, J. L. Thibault, G. Andersen, J. Pariset, A. Marcone, R. J. M. Lane, A. Hofman, M. Verin, T. Matilla, P. Bedoucha, J. Roche, M. Lai, M. Collard, A. Ugarte, F. Gallecho, D. Silbersweig, C. Kennard, J. P. Azulay, T. W. Ho, P. L. I. Dellemijn, R. Girardello, F. Baas, B. Voss, F. Rozenberg, E. M. Brocker, V. Stanev, A. A. J. Soeterboek, A. Marra, A. Rey, E. Ertem, M. Sawradewicz-Rybak, J. De Keyser, P. Cavallari, F. Proust, Y. Chevalier, H. C. Hansen, D. Leys, C. A. Davie, K. Hoang-Xuan, C. Bairati, H. van Crevel, Thomas T. Warner, B. Bompais, A. Dobbeleir, T Campbell, C. Macko, C. J. M. Klijn, M. Dussallant, T. P. Berlit, W. Rozenbaum, M. J. van den Bent, W. A. Rocca, M. Muller, H. Hundemer, U. Zifko, M. Campera, F. Drislane, D. Ranoux, T. M. Kloss, Anil Kumar, I. Ruolt, C. Bargnani, B. Marescau, N. A. Losseff, S. Notermans, B. Kint, E. T. Burke, C. Aykut, J. Matias Guiu, P. Maquet, T. Drogendijk, M. Leone, K. von Ammon, M. Pepeliarska, C. Prados, L. DiGiamberardino, T. Logtenberg, G. Lenoir, I. Castaldo, Damhaut, M. Radionova, G. Sirabian, R. Navon, Giovanni Antonini, K. Al Moutaery, E. Chamas, R. Schönhuber, M. Giannini, B. Debilly, I. Labatut, H. Henon, J. A. Egido, M. Baudrimont, J. N. Lorenzo, J. E. C. Bromberg, R. Antonacci, J. J. Vilchez, T. Moulin, B. Rautenstrauss, Giovanni Meola, J. Noth, S Mammi, P. Laforet, F. Lopez, C. Gehring, S. Bort, G. Rancurel, D. Decamps, S. Kostadinova, Y. Shapira, B. Neundoerfer, D. Chavrot, M. Solimena, J. P. Salier, W. Deberdt, R. Hoff-Jörgensen, A. Messina, S. Meairs, G. Rosoklija, E. Nelis, I. Bertran, C. Ertekin, J. Lohmeyer, Mitermayer Galvao dos Reis, L. Calo, E. Maccagnano, A. P. Hays, J. Verlooy, M. G. Forno, T. Blanco, L. Bail, Gabriella Silvestri, J. Montero, F. Bertrand, R. T. Ghnassia, C. Besses, T. Sereghy, F. Shalit, G. Bogliun, S. Braghi, St. Baykouchev, C. Franke, A. Lasa, L. C. Archard, J. Kriebel, S. Shaunak, M. Nocito, Alexander Tsiskaridze, E. Manfredini, T. Seigal, David G. Gadian, M. Barlas, J. D. Degos, C. Seeber, J. Caemert, J. L. Mas, R. B. Pepinsky, M. G. D'Angelo, N. Baumann, S. Yorifuji, H. P. Endtz, M. A. Cassatella, R. A. C. Hughes, V. Golzi, A. Bittencourt, A. Ferreira, M. Sanson, C. Alper, M. Vermeulen, M. A. A. van Walderveen, E. Alexiou, C. H. Lucas, M. Fiorelli, Y. N. Debbink, R. Gil, S. Congia, T. Banerjee, J. M. Bouchard, A. N. Pinto, A. Ceballos-Baumann, G. Grollier, P. I. M. Schmitz, M. D. Catata, N. Lahat, N. S. Rao, P. Papathanasopoulos, J. Valls-Solé, D. Claus, G. Schroter, A. Castro, C. Videbaek, R. Martinez Dreke, A. D. Platts, M. Hermesl, A. C. PeÇanha-Martins, M. Cardoso Silva, P. Masnou, M. J. A. Tanner, Ch. Confavreux, B. Mishu, H. Rasmussen, L. Valenciano, Carlo Pozzilli, S. W. Li, V. Salzman, Y. Vashtang, Massimo Franceschi, M. Severo, G. Deuschl, S. Setien, G. Mariani, A. Protti, J. Castillo, M. J. B. Taphoorn, M. Frontali, I. Milonas, D. Decoq, J. A. Navarro, S. Castellvi-Pel, C. Ertikin, M. Urtasun, Y. Lajat, B. E. Kendall, E. Verdu, B. Gueguen, E. Boisen, R. Couderc, A Danek, JM Stevens, F. Nicoli, L. Feltri, M. L. Vazquez-Andre, J. A. Morgan-Hughes, L. D'Angelo, F. Y. Liew, L. F. Pascual, J. Patrignani Ochoa, Vittorio Martinelli, J. Cophignon, L. Zhang, S. Martin, J. F. Meder, H. C. Buschmann, L. Bertin, J. van Gijn, A. Barreiro, A. Cools, C. Leon, A. Berod, E. A. Anllo, E. Zanette, L. Petrov, R. Barona, B. Gallicchio, P. J. Cozzone, N. Diederich, G. Cancel, L. Schelosky, P. Orizaola, K. Yulug, S. Ozer, Valeria A. Sansone, B. Guiraud-Chaumeil, K. Voigt, P. Labauge, M. Eoli, J. Zhu, J. Aguirre, M. Ferrarini, B. Zyluk, E. Planas, A. Cadilha, C. Tortorella, H. Bismuth, C. E. Counsell, A. Laun, A. Ferlini, Rio J. Montalban, N. Biary, L. Becker, M. Fardeau, M. Poloni, V. M. S. de Bruin, C. Fornada, J. Barros, E. Ganzmann, E. Touze, D. Wallach, J. Peila, H. Fujimura, M. T. Iba-Zizen, G. Macchi, C. Villoslada, R. Gouider, Ph. Rondepierre, P. Grummich, P. Chiodi, C. Conte, M. Michels, P. Annunziata, G. Semana, C. Sommer, J. Vajsar, D. Zekin, J. Kulisevsky, David G. Munoz, B. Jacotot, M. Magoni, A. Luxen, T. Garcia-Silva, S. Di Cesare, Christophe Tzourio, M. Gomori, I. Picomell, L. Santoro, F. Villa, Giovanni Pennisi, T. Ribalta, J. M. Molto, L. Marzorati, P. Loiseau, F. Gemignani, A. Gironell, J. Wissel, A. Prusinski, F. Cailloux, P. Villanueva-Hemandez, P. Cozzone, T. Del Ser, J. Sans-Sabrafen, M. Zappia, P. W. A. Willems, G. Tchernia, D. Gardeur, R. Bauer, F. Palomo, H. Metz, S. Lamoureux, C. Chastang, I. Reinhard, A. Goldfarb, S. Harder, Jordi Río, C. Ozkara, E. Tekinsoy, P. Vontobell, J. De Recondo, M. Rabasa, L. Lacomblez, F. Boon, Dgt Thomas, V. Palma, Renato Mantegazza, A. Dervis, M. Nueckel, B. YalÇinerner, I. Duran, G. Dalla Volta, A. Zubimendi, J. Pinheiro, A. Marbini, Xavier Montalban, H. Wekerle, X. Pereira Monteino, F. Crespo, F. Koskas, N. Battistini, C. Ruiz, H. Offner, J. de Pommery, P. Kanovsky, J. Y. Barnett, J. Pardo, G. Tomei, R. Rene, H. M. Lokhorst, P. Thajeb, H. Bilgin, D. McGehee, R. Fahsold, L. Morgante, Katie Sidle, C. Delwaide, M. N. Diaye, P. H. Rice, A. Creange, C. Sabev, K. Stephan, K. WeilBenborn, G. Magnani, L. Grymonprez, F. Cardellach, M. Kaps, N. G. Meco, F. Vega, V. Bonifati, A. Desomer, M. Baldy-Moulinier, G. Kvale, F. J. Authier, B. Yegen, T. Ho, J. M. Rozet, E. A. Cabanis, L. Bruce, L. Ambrosoli, M. A. Petrella, M. Hernandez, P. Timmings, H. B. van der Worp, F. Mahieux, A. Urbano-Marquez, D. A. Krendel, A. A. Garcia, R. Divari, R. Michalowicz, M. R. Piedmonte, M. Bondavalli, M. Zanca, P. F. Ippel, Onofre Combarros, B. Tavitian, E. Hirsch, I. Anastasopoulos, A. Roses, A. Köhler, P. Vienna, V. Timmerman, P. Sergi, F. Cornelio, A. Di Pasquale, R. Verleger, S. Castellvirel, J. Proano, B. van Moll, F. Rubio, W. Hacke, I. Lavenu, L. Zetta, M. W. Tas, N. Bittmann, M. Bonamini, O. R. Hommes, V. Dousset, N. Afsar, S. Belal, R. R. Myers, J. Goes, Giuseppe Vita, E. Clementi, V. G. Karepov, M. Jueptner, A Vincent, P. Emmrich, Th. Heb, A. Caballo, J. Gallego, T. Mokrusch, C. Perla, L. Gebuhrer, O. Titlbach, Alessandro Prelle, A. Czlonkowska, M. Russo, D. Hadjiev, T. S. Chkhikvishvili, M. Oehlschlager, G. Becker, I. Günther, E. N. Stenager, J. Garcia Agundez, J. Casademont, J. Batlle, S. Podobnik-Sarkanji, C. Alonso-Villaverde, B. Delaguillaume, B. Genc, B. Mazoyer, A. Rodriguez-Al-barino, Ch. Hilger, B. Ferrero, R. Price, W. Grisold, L. Fuhry, D. Oulbani, D. Ewing, A. Petkov, W. Walther, A. Gokyigit, John Newsom-Davis, J. Tayot, D. Seliak, G. Pelliccioni, D. Campagne, K. Kessler, F. Boureau, D. Perani, J. P. N'Guyen, N. Tchalucova, B. A. Antin-Ozerkis, C. Lacroix, B. D. Aronovich, I. H. Jenkins, E. A. dos Reis, M. Hortells, H. M. Meinck, H. Ch. Buschmann, S. C. J. M. Jacobs, T. Wetter, P. Creissard, N. Martinez, J. Weidenfeldl, H. J. Sturenburg, G. Damlacik, V. Gracia, J. C. Turpin, A. Pou-Serradell, J. P. Vincent, T. Gagoshidze, U. Ozkutlu, M. McLeod, K. Siegfried, I. Tchaoussoglou, J. Hildebrand, S. Kowalska, M. C. Picot, G. Galardin, L. Crevits, F. Andreetta, S. Larumbe-Lobalde, G. de la Sierra, J. C. Alvarez-Cermeno, R. J. Seitz, P. L. Oey, L. Ptacek, A. M. J. Paans, A. Wirrwar, A. Schmied, J. Uilchez, H. Tounsi, D. Hipola, V. Avoledo, Y. Hirata, P. Vermersch, T. M. Aisonobe, J. Valls-SoIè, H. Staunton, J. Dichgans, R. Karabudak, I. Dones, G. Porta, E. Janssens, Maria Martinez, J. M. Fernandez-Real, R. Villagra, Y. Yoshino, C. Kabus, K. Schimrigk, I. Girard-Buttaz, F. Piccoli, F. Aichner, P. Zuchegna, S. M. Al Deeb, F. Bono, N. Busquets, A. Jobert, Patrizia Ciscato, M. Martin, L. Polman, S. Darbra, V. Le Cam-Duchez, F. Baldissera, B. Baykan-Kurt, D. Guez, M. Bratoeva, H. Matsui, M. Mila, H. Perron, L. Bjorge, G. Husby, Steven T. DeKosky, D. R. Cornblath, J. M. Gabriel, J. J. Poza, Y. Wu, A. Toscano, R. P. Kleyweg, J. Kuhnen, S. O. Confort-Gouny, A. Barcelo, A. M. Conti, C. Fiol, C. Steichen-Wiehn, J. Rodes, M. Cavenaile, C. Vedeler, M. Drlicek, C. Argentino, M. L. Peris, A. Cervello, A. Z. GinaÏ, S. Yancheva, D. Passingham, S. Aoba, D. L. 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Frigo, Leif Østergaard, J. L. Munoz Blanco, A. Cruz-Matinez, J. De Reuck, C. Theillet, T. Barroso, V. Oikonen, Florence Lebert, M. Kilinc, C. Cordon-Cardon, G. Stoll, E. Thiery, F. Pulcinelli, J. Solski, M. Schmiegelow, L. J. Polman, P. Fernandez-Calle, C. Wikkelso, M. Ben Hamida, M. Laska, E. Kott, W. Sulkowski, C. Lucas, N. M. Bornstein, D. Schmitz, M. W. Lammers, A. de Louw, R. J. S. Wise, P. A. van Darn, C. Antozzi, P. Villanueva, P. H. E. Hilkens, C. Constantin, W. Ricart, A. Wolf, M. Gamba, P. Maguire, Alessandro Padovani, B. M. Patten, Marie Sarazin, H. Ackermann, L. Durelli, S. Timsit, Sebastian Jander, B. W. Scheithauer, G. Demir, J. P. Neau, P. Barbanti, A. Brand, N. AraÇ, V. Fischer-Gagnepain, R. Marchioli, G. Serratrice, C. Maugard-Louboutin, G. T. Spencer, D. Lücke, G. Mainardi, K. Harmant Van Rijckevorsel, G. B. Creel, R. Manzanares, Francesco Fortunato, A. May, J. Workman, K. Johkura, E. Fernandez, Carlo Colosimo, L. Calliauw, L. Bet, Félix F. Cruz-Sánchez, M. Dhib, H. Meinardi, F. Carrara, J. Kuehnen, C. Peiro, H. Lassmann, K. Skovgaard Olsen, A. McDonald, L. Sciulli, A. Cobo, A. Monticelli, B. Conrad, J. Bagunya, J. Benitez, V. Desnizza, B. Dupont, O. Delrieu, D. Moraes, J. J. Heimans, F. Garcia Rio, M. Matsumto, A. Fernandez, R. Nermni, R. Chalmers, M. J. Marchau, F. Aguado, P. Velupillai, P. J. Martin, P. Tassan, V. Demarin, A. Engelien, T. Gerriets, Comar, J. L. Carrasco, J. P. Pruvo, A. Lopez de Munain, D. Pavitt, J. Alarcon, Chris H. Polman, B. Guldin, N. Yeni, Hartmut Brückmann, N. Wilczak, H. Szwed, R. Causaran, G. Kyriazis, M. E. Westarp, M. Gasparini, N. Pecora, J. M. Roda, E. Lang, V. Scaioli, David R. Fish, D. Caputo, O. Gratzl, R. Mercelis, A. Perretti, G. Steimetz, I. Link, C. Rigoletto, A. Catafau, G. Lucotte, M. Buti, G. Fagiolari, A. Piqueras, C. Godinot, J. C. Meurice, Erodriguez J. Dominigo, F. Lionnet, H. Grzelec, David J. Brooks, P. M. G. Munro, F. X. Weilbach, M. Maiwald, W. Split, B. Widjaja-Cramer, V. Ozturk, J. Colas, E. Brizioli, J. Calleja, L. Publio, M. Desi, R. Soffietti, P. Cortinovis-Tourniaire, E. F. Gonano, G. Cavaletti, S. Uselli, K. Westerlind, H. Betuel, C. O. Dhiver, H. Guggenheim, M. Hamon, R. Fazio, P. Lehikoinen, A. Esser, B. Sadzot, G. Fink, Angelo Antonini, D. Bendahan, V. Di Carlo, G. Galardi, A. F. Boller, M. Aksenova, Del Fiore, V. de la Sayette, H. Chabriat, A. Nicoletti, A. Dilouya, M. L. Harpin, E. Rouillet, J. Stam, A. Wolters, M. R. Delgado, Eduardo Tolosa, G. Said, A. J. Lees, L. Rinaldi, A. Schulze-Bonhage, MA Ron, C. Lefebvre, E. W. Radü, R. Alvarez, M. L. Bots, P. Reganati, S. Palazzi, A. Poggi, N. J. Scolding, V. Sazdovitch, T. Moreau, E. Maes, M. A. Estelies, P. Petkova, Jose-Felix Marti-Masso, G De La Meilleure, N. Mullatti, M. Rodegher, N. C. Notermans, T. A. T. Warner, S. Aktan, J. P. Louboutin, L. Volpe, C. Scheidt, W. Aust, C. M. Wiles, U. Schneider, S. K. Braekken, W. R. Willems, K. Usuku, Peter M. Rothwell, C. Talamon, M. L. Sacchetti, A. Codina, M. H. Marion, A. Santoro, J. Roda, A. Bordoni, D. J. Taylor, S. Ertas, H. H. Emmen, J. Vichez, V. BesanÇon, R. E. Passingham, M. L. Malosio, A. Vérier, M. Bamberg, A. W. Hansen, E. Mostacero, G. Gaudriault, Marie Vidailhet, B. Birebent, K. Strijckmans, F. Giannini, T. Kammer, I. Araujo, J. Nowicki, E. Nikolov, A. Hutzelmann, R. Gherardi, J. Verroust, L. Austoni, A. Scheller, A. Vazquez, S. Matheron, H. Holthausen, J. M. Gerard, M. Bataillard, S. Dethy, V. H. Patterson, V. Ivanez, N. P. Hirsch, F. Ozer, M. Sutter, C. Jacomet, M. Mora, Bruno Colombo, A. Sarropoulos, T. H. Papapetropoulos, M. Schwarz, D. S. Dinner, N. Acarin, B. Iandolo, J. O. Riis, P. R. J. Barnes, F. Taroni, J. Kazenwadel, L. Torre, A. Lugaresi, I. L. Henriques, S. Pauli, S. Alfonso, Pedro Quesada, A. S. T. Planting, J. M. Castilla, Thomas Gasser, M. Van der Linden, A. Alfaro, E. Nobile-Orazio, G. Popova, W. Vaalburg, F. G. A. van der Mech, L. Williams, F. Medina, J. P. Vernant, J. Yaouanq, B. Storch-Hagenlocher, A. Potemkowski, R. Riva, M. H. Mahagne, M. Ozturk, Ve. Drory, N. Konic, C. Jungreis, A. Pou Serradell, J. L. Gauvrit, G. J. Chelune, S. Hermandez, T. Dingus, L. Hewer, Ch. Koch, M. N. Metz-Lutz, G. Parlato, M. Sinaki, Charles Pierrot-Deseilligny, H. C. Diener, J. Broeckx, J. Weill-Fulazza, M. L. Villar, M. Rizzo, O. Ganslandt, C. Duran, N. A. Fletcher, G. Di Giovacchino, Susan T. Iannaccone, C. Kolig, N. Fabre, H. A. Crockard, Rita Bella, M. Tazir, E. Papagiannuli, K. Overgaard, Emma Ciafaloni, I. Lorenzetti, F. Viader, P. A. H. Millac, I. Montiel, L. H. Visser, M. Palomar, P. L. Murgia, H. Pedersen, Rafael Blesa, S. Seddigh, W. O. Renier, I. Lemahieu, H. M. L. Jansen, L. Rosin, J. Galofre, K. Mattos, M. Pondal, G. M. Hadjigeorgiou, D. Francis, L. Cantin, D. Stegeman, M. Rango, A. B. M. F. Karim, S. Schraff, B. Castellotti, I. Iriarte, E. Laborde, T. J. Tjan, R. Mutani, D. Toni, B. Bergaasco, J. G. Young, C. Klotzsch, A. Zincone, X. Ducrocq, M. Uchuya, O. J. Kolar, A. Quattrone, T. Bauermann, Nereo Bresolin, J. Vallée, B. C. Jacobs, A. Campos, Werner Poewe, J. A. Villanueva, A. W. Kornhuber, A. Malafosse, E. Diez-Tejedor, G. Jungreia, M. J. A. Puchner, A. Komiyama, O. Saribas, V. Volpini, L. Geremia, S. Bressi, A. Nibbio, Timothy E. Bates, T. z. Tzonev, E. Ideman, G. A. Damlacik, G. Martino, G. Crepaldi, T. Martino, Kjell Någren, E. Idiman, D. Samuel, J. M. Perez Trullen, Y. van der Graaf, J. O. Thorell, M. J. M. Dupuis, E. Sieber, R. D'Alessandro, C. Cazzaniga, J. Faiss, A. Tanguy, A. Schick, I. Hoksergen, A. Cardozo, R. Shakarishvili, G. K. Wennlng, J. L. Marti-Vilalta, J. Weissenbach, I. L. Simone, Amalia C. Bruni, Darius J. Adams, C. Weiller, A. Pietrangeli, F. Croria, C. Vigo-Pelfrey, Patricia Limousin, A. Ducros, G. Conti, O. Lindvall, E. Richter, M. Zuffi, A. Nappo, T. Riise, J. Wijdenes, M. J. Fernandez, J. Rosell, P. Vermersh, S. Servidei, M. S. C. Verdugo, F. Gouttiere, W. Solbach, M. Malbezin, I. S. Watanabe, A. Tumac, W. I. McDonald, D. A. Butterfield, P. P. Costa, F. deRino, F. Bamonti, J. M. Cesar, C. H. Lahoz, I. Mosely, M. Starck, M. H. Lemaitre, K. M. Stephan, S. Tex, R. Bokonjic, I. Mollee, L. Pastena, M. Gutierrez, F. Boiler, M. C. Martinez-Para, M. Velicogna, O. Obuz, A. Grinspan, M. Guarino, L. M. Cartier, E. Ruiz, D. Gambi, S. Messina, M. Villa, Michael G. Hanna, J. Valk, Leone Pascual, M. Clanet, Z. Argov, B. Ryniewicz, E. Magni, B. Berlanga, K. S. Wong, C. Gellera, C. Prevost, F. Gonzalez-Huix, R. Petraroli, J. E. G. Benedikz, I. Kojder, C. Bommelaer, L. Perusse, M. R. Bangioanni, Guy M. McKhann, A. Molina, C. Fresquet, E. Sindern, Florence Pasquier, M. J. Rosas, M. Altieri, O. Simoncini, M. Koutroumanidis, C. A. F. Tulleken, M. Dary-Auriol, S. Oueslati, H. Kruyer, I. Nishisho, C. R. Horning, A. Vital, G. V. Czettritz, J. Ph. Neau, B. Mihout, A. Ameri, M. Francis, S. Quasthoff, D. Taussig, S. Blunt, P. Valentin, C. Y. Gao, O. Heinzlef, H. d'Allens, C. Coudero, M. Erfas, G. Borghero, P. J. Modrego Pardo, M. C. Patrosso, N. L. Gershfeld, P. A. J. M. Boon, O. Sabouraud, M. Lara, J. Svennevig, G. L. Lenzi, A. Barrio, H. Villaroya, JosÇ M. Manubens, O. Boespflug-Tanguy, M. Carreras, D. A. Costiga, J. P. Breux, S. Lynn, C. Oliveras Ley, A. G. Herbaut, J. Nos, C. Tornali, Y. A. Hekster, J. L. Chopard, J. M. Manubens, P. Chemouilli, A. Jovicic, F. Dworzak, S. Smirne, S. E. Soudain, B. Gallano, D. Lubach, G. Masullo, G. Izquierdo, A. Pascual Leone Pascual, A. Sessa, V. Freitas, O. Crambes, L. Ouss, G. W. Van Dijk, P. Marchettini, P. Confalonieri, M. Donaghy, A. Munnich, M. Corbo, and M. E. L. van der Burg

Subjects
Neurology, business.industry, Media studies, Library science, Medicine, Neurology (clinical), business
Published
1994
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7. Irish neurological association

Author

D. McEneaney, S. A. Hawkins, W. P. Gray, D. O’Brien, D. Q. Ryder, T. F. Buckley, B. Brankin, N. M. Hart, S. L. Cosby, Z. Fabry, M. Bailey, I. V. Allen, R. W. McVicker, O. E. P. Shanks, R. J. McClelland, W. I. Forsythe, P. Holland, R. Butler, P. Brok, J. Lamb, A. Halaka, T. Burke, D. McMackin, H. Staunton, J. Phillips, M. Reilly, M. Hutchinson, T. Esmonde, P. J. Morrison, N. C. Nevin, W. P. Johnston, S. F. Refsum, I. C. Bailey, B. G. Mathew, J. I. Morrow, M. W. Swallow, M. Gibson, R. K. Vasishta, M. Mirakhur, S. Cameron, B. Sharma, M. Hally, C. Keohane, D. Ryder, M. Watt, W. J. Gray, B. Mathew, D. Bounds, Victoria Wood, V. Bhandari, D. P. Nicholls, C. G. H. West, N. J. Gutowski, R. P. Murphy, P. Buckley, A. Freyne, A. McCarthy, C. Larkin, C. H. S. Cameron, V. H. Patterson, E. Hicks, V. Patterson, and P. Crean

Subjects
medicine.medical_specialty, Irish, business.industry, Family medicine, language, Medicine, Optometry, General Medicine, business, language.human_language
Published
1993
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8. The vegetative state

Author

H, Staunton

Subjects
Social Identification, Incidence, Persistent Vegetative State, Humans, Electroencephalography, Ireland
Published
2009

9. PREFACE

Author

H. Staunton

Subjects
Literature, History, business.industry, Literary magazine, Literary science, Literary criticism, business
Published
2009
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10. Evaluation of the 2007 Toyota Camry Hybrid Synergy Drive System

Author

Joseph Philip Cunningham, Robert H Staunton, Timothy A. Burress, Larry E. Seiber, Chester Coomer, Steven Campbell, and Laura D. Marlino

Subjects
Electric motor, Engineering, business.product_category, business.industry, Automotive industry, Propulsion, Automotive engineering, Work (electrical), Power electronics, Electric vehicle, Systems engineering, Fuel efficiency, business, Efficient energy use
Abstract

The U.S. Department of Energy (DOE) and American automotive manufacturers General Motors, Ford, and DaimlerChrysler began a five-year, cost-shared partnership in 1993. Currently, hybrid electric vehicle (HEV) research and development is conducted by DOE through its FreedomCAR and Vehicle Technologies (FCVT) program. The mission of the FCVT program is to develop more energy efficient and environmentally friendly highway transportation technologies. Program activities include research, development, demonstration, testing, technology validation, and technology transfer. These activities are aimed at developing technologies that can be domestically produced in a clean and cost-competitive manner. Under the FCVT program, support is provided through a three-phase approach [1] which is intended to: • Identify overall propulsion and vehicle-related needs by analyzing programmatic goals and reviewing industry’s recommendations and requirements, then develop the appropriate technical targets for systems, subsystems, and component research and development activities; • Develop and validate individual subsystems and components, including electric motors, emission control devices, battery systems, power electronics, accessories, and devices to reduce parasitic losses; and • Determine how well the components and subassemblies work together in a vehicle environment or as a complete propulsion system and whether the efficiency and performance targets at the vehicle level have been achieved. The research performed in this area will help remove technical and cost barriers to enable technology for use in such advanced vehicles as hybrid electric, plug-in hybrid electric, electric, and fuel-cell-powered vehicles.

Published
2008
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11. The inheritance of MS susceptibility

Author

Anthony G. O'Farrell, H. Staunton, E. Keelan, and D. Lord

Subjects
Male, Genetics, Multiple Sclerosis, Models, Genetic, genetic structures, business.industry, Multiple sclerosis, General Medicine, medicine.disease, Object (computer science), Inheritance (object-oriented programming), Risk Factors, medicine, Humans, Female, Optic neuritis, Disease Susceptibility, Prevalence ratio, business
Abstract

The object of this study was to determine, if possible, the mode of inheritance of the susceptibility to multiple sclerosis (MS). It was known that no single-gene model could fit the observations, so oligogenic models (models involving a small number of genes) were sought. Oligogenic hypotheses were tested against the available population data for MS until a reasonable level of agreement was found. The best-fitting simple hypothesis was this: MS occurs only in people who are homozygous for a recessive gene and carry a dominant X gene, and then only with reduced penetrance. The dangerous allele m- at the autosomal locus appears to be fairly uniformly distributed across England, Ireland and Canada, occurring in 10-30% of the gene pool. There are large variations in the frequency of the allele s- at the X-locus, ranging from 10% up to 72% of the gene pool. The penetrance varies significantly with geographical location, but nowhere approaches unity, so that the environmental factors are of great importance. The hypothesis explains the broad features of the population pattern of the occurrence of MS and it gives an outstanding fit to the best available data on MS in relatives. The result may assist attempts to map the genetic data on MS, opens the way for a reassessment of the attempts to identify the environmental factors, and it makes possible the completion of nonempirical risk tables for various countries. Similar techniques may be applied to other disorders with a genetic component in their aetiology.

Published
1990
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12. Sudeck atrophy

Author

H, Staunton

Subjects
Reflex Sympathetic Dystrophy, Humans, History, 20th Century, Prognosis
Abstract

This paper reviews the contribution of Sudeck to the understanding of the condition commonly referred to as 'Sudeck's atrophy' and which is commonly used as a synonym for a condition variously called reflex sympathetic dystrophy, causalgia, algodystrophy and others. Sudeck came to show in his later papers that the so-called atrophy was, in the majority of cases, a normal inflammatory process of bone change in the course of healing after an inflammatory/infective or traumatic insult. Contrary to the views of much current literature, the vast majority of such cases had a good prognosis. In those cases which became pathological and had a correspondingly poorer prognosis, the characteristic clinical picture becomes associated with radiological and pathological changes, which, uniquely, are described by Sudeck. A knowledge of such radiological and pathological substrate for clinical symptomatology is important in the analysis of pain following trauma.

Published
2007

13. Barriers to the Application of High-Temperature Coolants in Hybrid Electric Vehicles

Author

Michael Starke, John S Hsu, and Robert H Staunton

Subjects
Electric motor, Engineering, Power rating, business.industry, Engine efficiency, Mechanical engineering, Internal combustion engine cooling, Electronics, business, Automotive engineering, Manufacturing cost, Traction motor, Coolant
Abstract

This study was performed by the Oak Ridge National Laboratory (ORNL) to identify practical approaches, technical barriers, and cost impacts to achieving high-temperature coolant operation for certain traction drive subassemblies and components of hybrid electric vehicles (HEV). HEVs are unique in their need for the cooling of certain dedicated-traction drive subassemblies/components that include the electric motor(s), generators(s), inverter, dc converter (where applicable), and dc-link capacitors. The new coolant system under study would abandon the dedicated 65 C coolant loop, such as used in the Prius, and instead rely on the 105 C engine cooling loop. This assessment is important because automotive manufacturers are interested in utilizing the existing water/glycol engine cooling loop to cool the HEV subassemblies in order to eliminate an additional coolant loop with its associated reliability, space, and cost requirements. In addition, the cooling of power electronic devices, traction motors, and generators is critical in meeting the U.S. Department of Energy (DOE) FreedomCAR and Vehicle Technology (FCVT) goals for power rating, volume, weight, efficiency, reliability, and cost. All of these have been addressed in this study. Because there is high interest by the original equipment manufacturers (OEMs) in reducing manufacturing cost to enhance their competitive standing, themore » approach taken in this analysis was designed to be a positive 'can-do' approach that would be most successful in demonstrating the potential or opportunity of relying entirely on a high-temperature coolant system. Nevertheless, it proved to be clearly evident that a few formidable technical and cost barriers exist and no effective approach for mitigating the barriers was evident in the near term. Based on comprehensive thermal tests of the Prius reported by ORNL in 2005 [1], the continuous ratings at base speed (1200 rpm) with different coolant temperatures were projected from test data at 900 rpm. They are approximately 15 kW with 103 C coolant and 20 kW with 50 C coolant. To avoid this 25% drop1 in continuous power, design changes for improved heat dissipation and carefully managed changes in allowable thermal limits would be required in the hybrid subsystems. This study is designed to identify the technical barriers that potentially exist in moving to a high-temperature cooling loop prior to addressing the actual detailed design. For operation at a significantly higher coolant temperature, there were component-level issues that had to be addressed in this study. These issues generally pertained to the cost and reliability of existing or near term components that would be suitable for use with the 105 C coolant. The assessed components include power electronic devices/modules such as diodes and insulated-gate bipolar transistors (IGBTs), inverter-grade high-temperature capacitors, permanent magnets (PM), and motor-grade wire insulation. The need for potentially modifying/resizing subassemblies such as inverters, motors, and heat exchangers was also addressed in the study. In order to obtain pertinent information to assist ORNL researchers address the thermal issues at the component, module, subassembly, and system levels, pre-existing laboratory test data conducted at varying temperatures was analyzed in conjunction with information obtained from technical literature searches and industry sources.« less

Published
2006
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14. Evaluation of 2005 Honda Accord Hybrid Electric Drive System

Author

Laura D. Marlino, Timothy A. Burress, and Robert H Staunton

Subjects
Electric motor, Engineering, business.product_category, business.industry, Propulsion, Automotive engineering, Energy conservation, Work (electrical), Power electronics, Component (UML), Electric vehicle, Systems engineering, business, Efficient energy use
Abstract

The Hybrid Electric Vehicle (HEV) program officially began in 1993 as a five-year, cost-shared partnership between the U.S. Department of Energy (DOE) and American auto manufacturers: General Motors, Ford, and Daimler Chrysler. Currently, HEV research and development is conducted by DOE through its FreedomCAR and Vehicle Technologies (FCVT) program. The mission of the FCVT program is to develop more energy efficient and environmentally friendly highway transportation technologies. Program activities include research, development, demonstration, testing, technology validation, and technology transfer. These activities are aimed at developing technologies that can be domestically produced in a clean and cost-competitive manner. The vehicle systems technologies subprogram, which is one of four subprograms under the FCVT program, supports the efforts of the FreedomCAR through a three-phase approach [1] intended to: (1) Identify overall propulsion and vehicle-related needs by analyzing programmatic goals and reviewing industry's recommendations and requirements, then develop the appropriate technical targets for systems, subsystems, and component research and development activities; (2) Develop and validate individual subsystems and components, including electric motors, emission control devices, battery systems, power electronics, accessories, and devices to reduce parasitic losses; and (3) Determine how well the components and subassemblies work together in a vehicle environment or as a complete propulsion system and whether the efficiency and performance targets at the vehicle level have been achieved. The research performed under the vehicle systems subprogram will help remove technical and cost barriers to enable technology for use in such advanced vehicles as hybrid electric, plug-in electric, and fuel-cell-powered vehicles.

Published
2006
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15. Evaluation of 2004 Toyota Prius Hybrid Electric Drive System

Author

Laura D. Marlino, Curtis W. Ayers, Timothy A. Burress, Robert H Staunton, and John Chiasson

Subjects
Electric motor, Engineering, Rotor (electric), business.industry, Propulsion, Automotive engineering, law.invention, law, Derating, Range (aeronautics), Fuel efficiency, business, Efficient energy use, Petrol engine
Abstract

The 2004 Toyota Prius is a hybrid automobile equipped with a gasoline engine and a battery- and generator-powered electric motor. Both of these motive-power sources are capable of providing mechanical-drive power for the vehicle. The engine can deliver a peak-power output of 57 kilowatts (kW) at 5000 revolutions per minute (rpm) while the motor can deliver a peak-power output of 50 kW over the speed range of 1200-1540 rpm. Together, this engine-motor combination has a specified peak-power output of 82 kW at a vehicle speed of 85 kilometers per hour (km/h). In operation, the 2004 Prius exhibits superior fuel economy compared to conventionally powered automobiles. To acquire knowledge and thereby improve understanding of the propulsion technology used in the 2004 Prius, a full range of design characterization studies were conducted to evaluate the electrical and mechanical characteristics of the 2004 Prius and its hybrid electric drive system. These characterization studies included (1) a design review, (2) a packaging and fabrication assessment, (3) bench-top electrical tests, (4) back-electromotive force (emf) and locked rotor tests, (5) loss tests, (6) thermal tests at elevated temperatures, and most recently (7) full-design-range performance testing in a controlled laboratory environment. This final test effectively mapped the electrical and thermal results for motor/inverter operation over the full range of speeds and shaft loads that these assemblies are designed for in the Prius vehicle operations. This testing was undertaken by the Oak Ridge National Laboratory (ORNL) as part of the U.S. Department of Energy (DOE) - Energy Efficiency and Renewable Energy (EERE) FreedomCAR and Vehicle Technologies (FCVT) program through its vehicle systems technologies subprogram. The thermal tests at elevated temperatures were conducted late in 2004, and this report does not discuss this testing in detail. The thermal tests explored the derating of the Prius motor design if operated at temperatures as high as is normally encountered in a vehicle engine. The continuous ratings at base speed (1200 rpm) with different coolant temperatures are projected from test data at 900 rpm. A separate, comprehensive report on this thermal control study is available [1].

Published
2006
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16. Microturbine Power Conversion Technology Review

Author

R. H. Staunton and B. Ozpineci

Subjects
Engineering, Reliability (semiconductor), business.industry, Power electronics, Electrical engineering, Electronics, Microgrid, Voltage regulation, Voltage source, AC power, Converters, business, Reliability engineering
Abstract

In this study, the Oak Ridge National Laboratory (ORNL) is performing a technology review to assess the market for commercially available power electronic converters that can be used to connect microturbines to either the electric grid or local loads. The intent of the review is to facilitate an assessment of the present status of marketed power conversion technology to determine how versatile the designs are for potentially providing different services to the grid based on changes in market direction, new industry standards, and the critical needs of the local service provider. The project includes data gathering efforts and documentation of the state-of-the-art design approaches that are being used by microturbine manufacturers in their power conversion electronics development and refinement. This project task entails a review of power converters used in microturbines sized between 20 kW and 1 MW. The power converters permit microturbine generators, with their non-synchronous, high frequency output, to interface with the grid or local loads. The power converters produce 50- to 60-Hz power that can be used for local loads or, using interface electronics, synchronized for connection to the local feeder and/or microgrid. The power electronics enable operation in a stand-alone mode as a voltage source ormore » in grid-connect mode as a current source. Some microturbines are designed to automatically switch between the two modes. The information obtained in this data gathering effort will provide a basis for determining how close the microturbine industry is to providing services such as voltage regulation, combined control of both voltage and current, fast/seamless mode transfers, enhanced reliability, reduced cost converters, reactive power supply, power quality, and other ancillary services. Some power quality improvements will require the addition of storage devices; therefore, the task should also determine what must be done to enable the power conversion circuits to accept a varying dc voltage source. The study will also look at technical issues pertaining to the interconnection and coordinated/compatible operation of multiple microturbines. It is important to know today if modifications to provide improved operation and additional services will entail complete redesign, selected component changes, software modifications, or the addition of power storage devices. This project is designed to provide a strong technical foundation for determining present technical needs and identifying recommendations for future work.« less

Published
2003
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18. Neurofibromatosis: why not Smith's disease?

Author

H. Staunton and Lyons Jb

Subjects
medicine.medical_specialty, History, Neurofibromatoses, General Neuroscience, History, 19th Century, Disease, medicine.disease, Dermatology, History and Philosophy of Science, medicine, Humans, Neurology (clinical), Neurofibromatosis, Ireland
Published
1992

19. Irish (Donegal) amyloidosis is associated with the transthyretinALA60 (Appalachian) variant

Author

H. Staunton, R. J. Guiloff, N. Miyazato, A E Harding, M. Nakazato, and M. B. Davis

Subjects
Male, medicine.medical_specialty, Molecular Sequence Data, Cardiac Output, Low, Late onset, Northern Ireland, Northern ireland, Transthyretin Gene, Irish, Appalachian Region, Internal medicine, medicine, Humans, Prealbumin, Symptom onset, Amino Acid Sequence, Aged, Genetics, biology, business.industry, Amyloidosis, nutritional and metabolic diseases, Peripheral Nervous System Diseases, DNA, medicine.disease, language.human_language, Transthyretin, Endocrinology, Mutation, language, biology.protein, Female, Neurology (clinical), business
Abstract

A cluster of cases of late onset amyloidosis, presenting chiefly with peripheral neuropathy and cardiac failure, has been described in Donegal, north-west Ireland. Molecular genetic and protein analyses show that two new patients from the same region have a mutation in the transthyretin gene previously reported in a family with an Irish ancestor from the Appalachian region of the United States.

Published
1991

20. Anginal Headache and Its Basis

Author

J Horgan, A Grace, K Breathnach, and H Staunton

Subjects
Male, medicine.medical_specialty, business.industry, medicine.medical_treatment, Headache, Myocardial Ischemia, General Medicine, Middle Aged, medicine.disease, Angina, Bypass surgery, Occipital headache, Anesthesia, Angioplasty, Internal medicine, medicine, Cardiology, Humans, Neurology (clinical), business, Chest pain angina
Abstract

A case is presented of angina manifesting itself initially solely as vertex and occipital headache, accompanied by EKG changes, provoked by exercise and relieved by rest. It was totally relieved by coronary bypass surgery and, later, by angioplasty. Its mechanism is probably a variation on the neural convergence usually invoked to explain the more typical chest pain angina.

Published
1997
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21. Pregnancy, hormones and epilepsy

Author

J. Stronge, H. Staunton, J. F. Stratton, and P. Bosio

Subjects
Epilepsy, Pregnancy hormones, business.industry, medicine, Obstetrics and Gynecology, Physiology, medicine.disease, business
Published
1995
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23. Proceedings of the Irish Neurological Association 24th Annual Scientific Meeting, Beaumont Hospital, May 1988

Author

J. A. O’Dwyer, J. A. Toland, M. Geraghty, O. Hardiman, M. Keogan, B. O’Moore, M. King, M. A. Farrell, D. O’Neill, M. Rowan, D. Abrahamson, J. P. Walsh, D. Coakley, J. Feeley, J. Fahy, D. Glynn, M. Hutchinson, J. McMenamin, G. Monaghan, Y. Khan, T. Diamond, W. J. Gray, C. P. Chee, T. F. Fannin, S. F. Murphy, J. P. Phillips, S. Connolly, Dermot P. Byrnes, V. Patterson, E. Hicks, T. Taylor, R. H. Brown, J. J. Halperin, B. E. Shapiro, I. S. Wray, D. McMackin, S. Murphy, H. Staunton, J. Phillips, M. Farrell, I. Radford, K. J. Trew, S. A. Hawkins, T. E. Burke, T. Keelin, D. Lord, A. G. O’Farrell, M. J. Connolly, J. Stack, E. A. Martin, A. Bergin, C. Keoghane, N. Callaghan, T. O’Riordan, P. A. Daly, A. Shattock, S. Gardner, M. G. Davies, M. J. Rowan, P. MacMathuna, P. W. N. Keeling, D. G. Weir, J. Feely, P. McLoughlin, T. Keelan, W. Tormey, J. Donohoe, C. O’Donovan, O. Browne, J. J. Dinn, G. C. Fry, C. N. Pidgeon, M. Regan, J. Moran, Liz Moran, R. O’Kennedy, and G. Kaar

Subjects
medicine.medical_specialty, Irish, business.industry, Family medicine, Ophthalmology, Association (object-oriented programming), language, Medicine, General Medicine, business, language.human_language
Published
1989
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24. Irish Neurological Association Proceedings of the 23rd Annual Scientific Meeting of the Irish Neurological Association — Galway, 9th–10th October 1987

Author

G. Dean, J. Cooney, W. J. Gray, R. H. S. Thompson, Brian E. Leonard, C. Feighery, C. Gibbons, J. M. French, Michael Hutchinson, T. Goggin, M. McLaughlin, Cheryl McCusker, S. Price, V. Patterson, J. Bodansky, J. McKirgan, E. Campbell, D. Byrnes, R. McConnell, J. Christie, Alan J. Thompson, D. Bates, M. J. Rosner, A. D. Smith, T. Pullar, H. Gough, M. Crowley, P. Ng, R. Clarke, K. Hampton, S. Young, C. Keohane, Sara Louise Cosby, B. Morrissey, Michael Swallow, J. Feeley, R. Peatfield, S. McQuaid, Ingrid V. Allen, N. Callaghan, D. Abrahams, D. O’Neill, J. J. Dinn, M. Baker, Stanley Hawkins, M. Feely, M. Lucock, C. Walton, J. A. O’Dwyer, A. Saaid, N. E. F. Cartlidge, P. Ryan, Ian Forsythe, J. Toland, I. Graham, E. A. Martin, D. McCormick, M. J. Taylor, M. A. Farrell, J. Moran, N. Collins, A. Bissessar, D. McInerney, Kathryn Boyd, Michael J. Rowan, J. B. Lyons, R. Hartley, S. O’Laoire, Davis Coakley, S. J. Martin, M. J. Redmond, I. Wallace, J. P. Phillips, G. F. Kaar, E. Carmody, James Bernard Walsh, B. McLain, H. Dardouri, A. Garrett, T. A. McNeill, H. Staunton, J. Kirk, J. H. D. Millar, B. K. Rima, M. Bailey, I. Brazil, I. A. O’Dwyer, and M. J. T. FitzGerald

Subjects
medicine.medical_specialty, Irish, business.industry, Family medicine, Ophthalmology, language, medicine, General Medicine, business, language.human_language
Published
1988
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25. Neurological association Proceedings of the 18th annual meeting of the Irish Neurological Association held in Galway on 5th and 6th November, 1982

Author

P. Dervan, Peter O. Behan, Ian Forsythe, Michael G. Harrington, I. Sbeih, I. C. Bailey, D. P. McInerney, Jim Malone, Lorna Browne, N. Callaghan, P. Carey, Stephanie Blandford, Michael Hutchinson, K. P. Maher, H. Staunton, Bernadette Horner, C. Niamatali, S. A. O’Laoire, N. MacDermott, Peter J. Kelly, J. Keating, J. H. Moran, Elodie Martin, M. H. R. Hutchinson, M. A.R.K. McCormick, B. O’moore, G. D. Hurley, Aidan Twomey, Alan J. Thompson, D. P. MacErlean, John A. O’Dwyer, J. Phillips, R. A. Johnston, M. Feely, Bryan J O’Neill, and A. P. McGeorge

Subjects
medicine.medical_specialty, Irish, business.industry, Family medicine, language, medicine, Physiology, General Medicine, business, language.human_language
Published
1983
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27. Hereditary amyloid polyneuropathy in north west Ireland

Author

Peter Dervan, R. Kale, H. Staunton, Reinhold P. Linke, and Peter Kelly

Subjects
Male, Pathology, medicine.medical_specialty, Autonomic Nervous System, Nerve conduction velocity, HLA Antigens, medicine, Humans, Aged, Nerve biopsy, biology, medicine.diagnostic_test, business.industry, Amyloidosis, Familial amyloid neuropathy, Middle Aged, medicine.disease, Immunohistochemistry, Pedigree, Amyloid Neuropathy, Transthyretin, biology.protein, Female, Neurology (clinical), Age of onset, Nervous System Diseases, business, Polyneuropathy, Ireland
Abstract

Seven cases of chronic sensorimotor polyneuropathy due to amyloidosis, from 7 different families, are described, in addition to the pathology in a sibling of 1 case. The age of onset ranged from 55 to 72 years. Cardiac involvement, intermittent diarrhoea and syncopal attacks were a frequent occurrence. Motor conduction velocity showed a moderate degree of slowing in 5 of 6 cases studied and marked slowing in 1. Amyloid deposits were seen in nerve biopsy material of all 8 subjects and in rectal mucosa from 1. Immunohistochemical identification revealed AF (transthyretin-derived)--amyloid in all 8 instances, confirming the presence of type 1 familial amyloid neuropathy. The genealogical data supported this analysis. Six of the 8 cases originated in a small area of the north-west coast of County Donegal in Ireland. The remaining cases also originated in the same county.

Published
1987

28. Polio revisited

Author

P, Murphy, H, Staunton, and R, Hone

Subjects
Adult, Male, Poliovirus Vaccine, Inactivated, Humans, Poliomyelitis
Published
1982

30. Anticonvulsants

Author

H, Staunton

Subjects
Epilepsy, Humans, Anticonvulsants
Published
1982

32. Cellular Immune Function to Measles Antigen in Normal Subjects and Multiple Sclerosis: Skin Testing with Autologous CSF and Other Antigens in Multiple Sclerosis

Author

M. Robinson, J. Dempsey, V. Parameswaran, L. Bannon, and H. Staunton

Subjects
Diminution, Pathology, medicine.medical_specialty, business.industry, Multiple sclerosis, medicine.disease, Measles, In vitro, Immune system, Cerebrospinal fluid, Antigen, In vivo, Immunology, medicine, business
Abstract

There have been reports of generally depressed cellular immune function in multiple sclerosis (1, 4). Reference has been made to a selective diminution in the cellular immune response to measles antigen (3, 2). With a view to testing the latter hypothesis and, if confirming it, reversing the situation by administration of transfer factor from unaffected siblings, we instituted a study of cellular immune function to measles antigen particularly in normal individuals and in subjects with multiple sclerosis. In addition, in view of the many criticisms of in vitro tests as an index of cellular immune capacity, an in vivo investigation on the basis of skin testing, with autologous cerebrospinal fluid and a variety of antigens was also undertaken.

Published
1979
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33. Review of 36 cases with extra-cranial arterial occlusion

Author

W, MacGowan, G, Lynch, and H, Staunton

Subjects
Adult, Carotid Artery Diseases, Male, Arteriosclerosis, Angiography, Endarterectomy, Middle Aged, Aneurysm, Aortography, Blood Vessel Prosthesis, Postoperative Complications, Subclavian Steal Syndrome, Humans, Female, Aged
Published
1971

36. Confederate General James A. Walker soon after Civil War, ca. 1865-1866

Author

Burdette, J. H., Staunton, VA (photographer) and Burdette, J. H., Staunton, VA (photographer)

Abstract

Brig. Gen. James A. Walker (VMI Class of 1852) of the Stonewall Brigade, in civilian clothes shortly after the end of the Civil War., Original owned by and copy used with permission of Washington & Lee University Special Collections.

37. Royal Academy of Medicine in Ireland — Section of Ophthalmology Proceedings of meeting held on Friday, 4th December, 1987

Author

M. F. J. Armstrong, D. B. Archer, M. Hope-Ross, A. Benedict-Smith, M. Hillery, P. Mullaney, M. Hickey-Dwyer, L. M. T. Collum, B. Power, S. Travers, D. Mooney, E. Bonnar, P. Logan, P. Eustace, D. F. P. Larkin, P. O’Domhnaill, E. Burke, and H. Staunton

Subjects
medicine.medical_specialty, Diabetic macular oedema, business.industry, Ophthalmology, Section (typography), Pupil size, Medicine, Optometry, General Medicine, business
Published
1988
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38. Points to consider when initiating clinical investigations in autistic paediatric populations-A White Paper.

Author

Ham LM, Staunton H, Schulz JM, Tillmann J, Volz D, Murtagh L, Chatham C, O'Connor EC, Chamberlain S, Schoenenberger P, Pandina G, Wang P, Kas MJH, Arango C, and Murphy D

Subjects
Child, Humans, Clinical Trials as Topic methods, Drug Development methods, Autism Spectrum Disorder therapy
Abstract

Many individuals with autism spectrum disorder (ASD) experience various degrees of impairment in social interaction and communication, restricted, repetitive behaviours, interests/activities. These impairments make a significant contribution to poorer everyday adaptive functioning. Yet, there are no pharmacological therapies to effectively treat the core symptoms of ASD. Since symptoms of ASD likely emerge from a complex interplay of vulnerabilities, environmental factors and compensatory mechanisms during the early developmental period, pharmacological interventions arguably would have the greatest impact to improve long-term outcomes when implemented at a young age. It is essential therefore, that clinical development programmes of investigational drugs in ASD include the paediatric population early on in clinical trials. Such trials need to offer the prospect of direct benefit (PDB) for participants. In most cases in drug development this prospect is supported by evidence of efficacy in adults. However, the effectiveness of treatment approaches may be age-dependent, so that clinical trials in adults may not provide sufficient evidence for a PDB in children. In this white paper, we consolidate recommendations from regulatory guidelines, as well as advice from the Food and Drug Administration, USA (FDA) and the Committee for Human Medicinal Products (CHMP) consultations on various development programmes on: 1) elements to support a PDB to participants in early paediatric clinical trials in ASD, including single-gene neurodevelopment disorders, 2) aspects of study design to allow for a PDB. This white paper is intended to be complementary to existing regulatory guidelines in guiding industry and academic sponsors in their conduct of early paediatric clinical trials in ASD., Competing Interests: Declaration of competing interest L.M.H., J.M.S., J.T., D.V., L.M., C.C., E.C.O., S.C., P.S. are full-time employees of F. Hoffmann - La Roche Ltd, Basel, Switzerland. H.S. is a full-time employee of Roche Products Ltd, Welwyn Garden City, UK. G.P. is a full-time employee of Janssen Research & Development, LLC, Raritan, NJ, USA . P.W. is an employee of Clinical Research Associates, LLC, CT, USA. M.K. has received (non-related) research funding from Novartis during the researching and writing of the manuscript. C.A. has been a consultant to or has received honoraria or grants from Acadia, Abbot, Ambrosetti, AMGEN, AstraZeneca, Bristol-Myers Squibb, Dainippon Sumitomo Pharma, Forum, Gedeon Richter, Janssen-Cilag, Lundbeck, Merck, Otsuka, Pfizer, Roche, Servier, Shire, Schering-Plough, Sunovion, and Takeda. D.M. has received funding for a PhD studentship from Compass, and for consulting from Jaguar Therapeutics and Hoffman La Roche., (Copyright © 2024. Published by Elsevier B.V.)

Published
2024
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39. The Clinical Development of Taldefgrobep Alfa: An Anti-Myostatin Adnectin for the Treatment of Duchenne Muscular Dystrophy.

Author

Muntoni F, Byrne BJ, McMillan HJ, Ryan MM, Wong BL, Dukart J, Bansal A, Cosson V, Dreghici R, Guridi M, Rabbia M, Staunton H, Tirucherai GS, Yen K, Yuan X, and Wagner KR

Abstract

Introduction: Duchenne muscular dystrophy (DMD) is a genetic muscle disorder that manifests during early childhood and is ultimately fatal. Recently approved treatments targeting the genetic cause of DMD are limited to specific subpopulations of patients, highlighting the need for therapies with wider applications. Pharmacologic inhibition of myostatin, an endogenous inhibitor of muscle growth produced almost exclusively in skeletal muscle, has been shown to increase muscle mass in several species, including humans. Taldefgrobep alfa is an anti-myostatin recombinant protein engineered to bind to and block myostatin signaling. Preclinical studies of taldefgrobep alfa demonstrated significant decreases in myostatin and increased lower limb volume in three animal species, including dystrophic mice., Methods: This manuscript reports the cumulative data from three separate clinical trials of taldefgrobep alfa in DMD: a phase 1 study in healthy adult volunteers (NCT02145234), and two randomized, double-blind, placebo-controlled studies in ambulatory boys with DMD-a phase 1b/2 trial assessing safety (NCT02515669) and a phase 2/3 trial including the North Star Ambulatory Assessment (NSAA) as the primary endpoint (NCT03039686)., Results: In healthy adult volunteers, taldefgrobep alfa was generally well tolerated and resulted in a significant increase in thigh muscle volume. Treatment with taldefgrobep alfa was associated with robust dose-dependent suppression of free myostatin. In the phase 1b/2 trial, myostatin suppression was associated with a positive effect on lean body mass, though effects on muscle mass were modest. The phase 2/3 trial found that the effects of treatment did not meet the primary endpoint pre-specified futility analysis threshold (change from baseline of ≥ 1.5 points on the NSAA total score)., Conclusions: The futility analysis demonstrated that taldefgrobep alfa did not result in functional change for boys with DMD. The program was subsequently terminated in 2019. Overall, there were no safety concerns, and no patients were withdrawn from treatment as a result of treatment-related adverse events or serious adverse events., Trial Registration: NCT02145234, NCT02515669, NCT03039686., (© 2024. The Author(s).)

Published
2024
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40. Stride Velocity 95th Centile Detects Decline in Ambulatory Function Over Shorter Intervals than the 6-Minute Walk Test or North Star Ambulatory Assessment in Duchenne Muscular Dystrophy.

Author

Rabbia M, Guridi Ormazabal M, Staunton H, Veenstra K, Eggenspieler D, Annoussamy M, Servais L, and Strijbos P

Subjects
Humans, Child, Male, Outcome Assessment, Health Care, Wearable Electronic Devices, Female, Muscular Dystrophy, Duchenne physiopathology, Muscular Dystrophy, Duchenne drug therapy, Walk Test, Walking physiology
Abstract

Background: Stride Velocity 95th Centile (SV95C) is the first wearable device-derived clinical outcome assessment (COA) to receive European Medicines Agency (EMA) qualification as a primary endpoint in ambulant patients with Duchenne muscular dystrophy (DMD) aged ≥4 years., Objective: To compare SV95C-in its first-ever clinical trial application as a secondary endpoint-with established motor function COAs used in the trial (Four-Stair Climb [4SC] velocity, North Star Ambulatory Assessment [NSAA], and Six-Minute Walk Distance [6MWD])., Methods: SV95C was a secondary endpoint in a subset (n = 47) of participants in the SPITFIRE/WN40227 trial of taldefgrobep alfa, which was discontinued due to lack of clinical benefit. Participants in the ≤48-week SV95C sub-study were 6-11 years old and received corticosteroids for ≥6 months pre-treatment. Pearson correlations were used to compare SV95C with the other COAs. Responsiveness and changes over time were respectively assessed via standardized response means (SRMs) based on absolute changes and mixed models for repeated measures., Results: SV95C change at Week 24 was -0.07 m/s, with limited variability (standard deviation: 0.16, n = 27). The SRM for SV95C indicated moderate responsiveness to clinical change at the earliest timepoint (Week 12, n = 46), while those of the other COAs did not indicate moderate responsiveness until Week 36 (6MWD, n = 33) or Week 48 (4SC velocity, n = 20; NSAA total score, n = 20). Baseline correlations between SV95C and other COAs were strong (r = 0.611-0.695). Correlations between SV95C change from baseline to Week 48 and changes in other COAs were moderate to strong (r = 0.443-0.678).∥., Conclusions: Overall, SV95C demonstrated sensitivity to ambulatory decline over short intervals, low variability, and correlation with established COAs. Although the negative trial precluded demonstration of SV95C's sensitivity to drug effect, these findings support the continued use of SV95C in DMD clinical trials.

Published
2024
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41. Two-year efficacy and safety of risdiplam in patients with type 2 or non-ambulant type 3 spinal muscular atrophy (SMA).

Author

Oskoui M, Day JW, Deconinck N, Mazzone ES, Nascimento A, Saito K, Vuillerot C, Baranello G, Goemans N, Kirschner J, Kostera-Pruszczyk A, Servais L, Papp G, Gorni K, Kletzl H, Martin C, McIver T, Scalco RS, Staunton H, Yeung WY, Fontoura P, and Mercuri E

Subjects
Humans, Pyrimidines adverse effects, Azo Compounds adverse effects, Spinal Muscular Atrophies of Childhood drug therapy, Muscular Atrophy, Spinal genetics
Abstract

Risdiplam is an oral, survival of motor neuron 2 (SMN2) pre-mRNA splicing modifier approved for the treatment of spinal muscular atrophy (SMA). SUNFISH (NCT02908685) Part 2, a Phase 3, randomized, double-blind, placebo-controlled study, investigated the efficacy and safety of risdiplam in type 2 and non‑ambulant type 3 SMA. The primary endpoint was met: a significantly greater change from baseline in 32-item Motor Function Measure (MFM32) total score was observed with risdiplam compared with placebo at month 12. After 12 months, all participants received risdiplam while preserving initial treatment blinding. We report 24-month efficacy and safety results in this population. Month 24 exploratory endpoints included change from baseline in MFM32 and safety. MFM‑derived results were compared with an external comparator. At month 24 of risdiplam treatment, 32% of patients demonstrated improvement (a change of ≥ 3) from baseline in MFM32 total score; 58% showed stabilization (a change of ≥ 0). Compared with an external comparator, a treatment difference of 3.12 (95% confidence interval [CI] 1.67-4.57) in favor of risdiplam was observed in MFM-derived scores. Overall, gains in motor function at month 12 were maintained or improved upon at month 24. In patients initially receiving placebo, MFM32 remained stable compared with baseline (0.31 [95% CI - 0.65 to 1.28]) after 12 months of risdiplam; 16% of patients improved their score and 59% exhibited stabilization. The safety profile after 24 months was consistent with that observed after 12 months. Risdiplam over 24 months resulted in further improvement or stabilization in motor function, confirming the benefit of longer-term treatment., (© 2023. The Author(s).)

Published
2023
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43. Home-based video assessment of ease of movement for patients with Duchenne: Interviews with physical therapists to select movement tasks.

Author

Contesse MG, Hodges J, Staunton H, Lowes LP, Elmankabadi B, Elder SJ, Ormazabal MG, Dalle Pazze L, and Leffler MG

Abstract

Background and Purpose: Patients with Duchenne muscular dystrophy (DMD) change their movement patterns to compensate for muscle weakness. The Duchenne Video Assessment (DVA) measures ease of movement through evaluation of compensatory movements. The DVA directs caregivers to video record patients performing home-based movement tasks using a mobile application, and DVA-certified physical therapists evaluate the videos using scorecards with prespecified compensatory movement criteria. Two qualitative interview studies were conducted to select movement tasks for the DVA that are relevant to patients with DMD and able to reflect changes in function., Methods: Qualitative interviews with eligible physical therapists were conducted remotely. Physical therapists were asked to spontaneously suggest movement tasks prior to evaluating specific movement tasks selected a priori. Analysts conducted a content analysis to investigate whether movement tasks selected a priori were confirmed as relevant to the population of interest and able to show changes in function., Results: The studies included five physical therapists to select tasks for ambulatory patients with DMD and six for non-ambulatory patients. For an ambulatory population, all five experts confirmed Climb Five Stairs, Run, Stand Up from Sitting, Sit Up from Supine, and Jump Forward, and four (80%) confirmed Walk as relevant and able to show functional changes. For a non-ambulatory population, all six experts confirmed Eat 10 Bites, Roll Over in Bed, Shift Weight in Bed, Take T-Shirt Off, Put T-Shirt On, Put Arms on Armrest, and Reach Across Table to Grab Cell Phone, and five (83%) confirmed Raise Hands Above Head as relevant and able to show functional changes., Discussion: Physical therapists confirmed the DVA movement tasks as relevant to patients with DMD and able to reflect changes in function. The use of the DVA in clinical trials provides an opportunity to collect data not seen in clinic and reduce travel burden for families., (© 2023 John Wiley & Sons Ltd.)

Published
2023
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44. A Mixed-method Approach to Develop an Ambulatory Module of the SMA Independence Scale.

Author

Staunton H, Cleanthous S, Teodoro V, Barrett L, Braid J, Ewens B, Cano S, Baranello G, Kirschner J, Belter L, and Mayhew A

Subjects
Child, Humans, Walking, Caregivers, Psychometrics methods, Quality of Life psychology, Muscular Atrophy, Spinal diagnosis
Abstract

Background: Limited qualitative data exist on the symptoms and impacts of spinal muscular atrophy (SMA) experienced by ambulant individuals. An ambulant module of the SMA Independence Scale (SMAIS) was developed to quantify the assistance required to perform everyday mobility-related activities., Objective: The objective of this study was to develop a patient-centered module that provides key insights into what constitutes independence for ambulant and near-ambulant individuals with SMA., Methods: A stepwise, mixed-method approach was used. Semi-structured interviews were conducted in three waves with individuals with SMA and caregivers of children with SMA who were ambulant or near-ambulant (can walk ≥5 steps with support). Wave 1 interviews (n = 20) focused on concept elicitation. Wave 2 and 3 interviews (n = 15, both) involved completion and cognitive debriefing of items generated based on Wave 1 interviews. Therapeutic area experts were consulted throughout all key steps of the study. In particular, feedback was provided for item refinement and response option decisions. A macro-level preliminary, exploratory analysis, using Rasch Measurement Theory (RMT), provided insight on measurement properties., Results: Wave 1 resulted in 42 mobility and 11 instrumental activity of daily living (iADL) items. During Wave 2, participants defined independence as completing a task with supportive aids but without help from another person, leading to item refinement and modifications to the response scale. Lack of conceptual relevance and ceiling effects led to the removal of all iADL items after Wave 2, and 41 mobility items were tested in Wave 3. Final exploratory RMT and item refinement to reduce overlap led to a 27-item set related to mobility tasks., Conclusions: Our study provides preliminary support for using the 27-item SMAIS-Ambulatory Module for ambulant or near-ambulant individuals with SMA. Larger-scale analyses to further assess the psychometric properties of the scale are warranted.

Published
2023
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45. Asthma Daytime Symptom Diary (ADSD) and Asthma Nighttime Symptom Diary (ANSD): Measurement Properties of Novel Patient-Reported Symptom Measures.

Author

Gater A, Nelsen L, Coon CD, Eremenco S, O'Quinn S, Khan AH, Eckert L, Staunton H, Bonner N, Hall R, Krishnan JA, Stoloff S, Schatz M, Haughney J, and Coons SJ

Subjects
Adolescent, Adult, Humans, Patient Reported Outcome Measures, Psychometrics, Reproducibility of Results, Surveys and Questionnaires, Asthma diagnosis
Abstract

Background: The Asthma Daytime Symptom Diary (ADSD) and the Asthma Nighttime Symptom Diary (ANSD) were developed to meet the need for standardized patient-reported measures of asthma symptoms to assess treatment trial outcomes in adults and adolescents., Objective: To determine scoring and evaluate the measurement properties of the ADSD/ANSD., Methods: Adolescents (12-17 years) and adults (18+ years) with asthma completed draft 8-item electronic versions of the ADSD/ANSD for 10 days alongside the Adult Asthma Symptom Daily Scales (AASDS) and a Patient Global Impression of Severity (PGIS). Using classical and modern psychometric methods, initial analyses evaluated the performance of ADSD/ANSD items to inform scoring. Subsequent analyses evaluated the reliability and validity of ADSD/ANSD scores., Results: A demographically and clinically diverse sample (n = 130 adolescents; n = 89 adults) was recruited. Item performance was generally strong. However, items assessing chest pressure and mucus/phlegm demonstrated redundancy and poorer performance and were removed. Principal-components analysis, confirmatory factor analysis, and item response theory supported combining items to form 6-item total ADSD/ANSD scores. Internal consistency (α = 0.94-0.95) and test-retest reliability (intraclass correlation coefficient = 0.86-0.95) were strong. Strong correlations (r = 0.72-0.80) were observed between ADSD scores and AASDS items assessing asthma symptom frequency, bother, and impact on activities. Significant differences (P < .001) in mean ADSD/ANSD scores were observed between groups categorized by asthma severity (PGIS), asthma control, inhaler use, nebulizer use, activity limitations, and nighttime awakenings., Conclusions: The ADSD/ANSD items and scores demonstrated strong reliability and validity. Implementation of the measures in interventional studies will enable the evaluation of responsiveness and meaningful within-patient change., (Copyright © 2021 The Authors. Published by Elsevier Inc. All rights reserved.)

Published
2022
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46. A Patient-Centered Evaluation of Meaningful Change on the 32-Item Motor Function Measure in Spinal Muscular Atrophy Using Qualitative and Quantitative Data.

Author

Duong T, Staunton H, Braid J, Barriere A, Trzaskoma B, Gao L, Willgoss T, Cruz R, Gusset N, Gorni K, Randhawa S, Yang L, and Vuillerot C

Abstract

The 32-item Motor Function Measure (MFM32) is an assessment of motor function used to evaluate fine and gross motor ability in patients with neuromuscular disorders, including spinal muscular atrophy (SMA). Reliability and validity of the MFM32 have been documented in individuals with SMA. Through semi-structured qualitative interviews ( N = 40) and an online survey in eight countries ( N = 217) with individuals with Types 2 and 3 SMA aged 2-59 years old and caregivers, the meaning of changes on a patient-friendly version of the MFM32 was explored. In an independent analysis of clinical trial data, anchor- and distribution-based analyses were conducted in a sample of individuals with Type 2 and non-ambulant Type 3 SMA to estimate patient-centered quantitative MFM32 meaningful change thresholds. The results from this study demonstrate that, based on patient and caregiver insights, maintaining functional ability as assessed by a patient-friendly version of the MFM32 is an important outcome. Quantitative analyses using multiple anchors (median age range of 5-8 years old across anchor groups) indicated that an ~3-point improvement in MFM32 total score represents meaningful change at the individual patient level. Overall, the qualitative and quantitative findings from this study support the importance of examining a range of meaningful change thresholds on the MFM32 including ≥0 points change reflecting stabilization or improvement and ≥3 points change reflecting a higher threshold of improvement. Future research is needed to explore quantitative differences in meaningful change on the MFM32 based on age and functional subgroups., Competing Interests: HS, JB, and TW are employees and shareholders of Roche Products Ltd. KG is an employee and shareholder of F. Hoffmann-La Roche Ltd. BT is an employee and shareholder of Genentech. TD serves on advisory boards and receives consultancy fees for Roche, Genentech, Biogen, Novartis and Cure SMA. CV is a PI for Roche clinical trials and has received consultancy fees from Roche, Biogen, and Avexis. TD, CV, and AB received consultancy fees from Roche for this project. NG is a volunteer of SMA Europe and SMA Schweiz, mother of a child living with SMA advisor and lecturer for Novartis Gene Therapies (AveXis) Biogen, Novartis, and Roche. RC was an employee with Cure SMA at the time of the study. LG is an employee of Analystat Corporation working in US Medical Affairs at Genentech and Roche as a statistical consultant and has received payment from Genentech Inc. SR is an employee of Adelphi Values, a health outcomes research agency, commissioned and paid by Roche to conduct the qualitative interview part of the study. LY is an employee of Charles River Associates, commissioned and paid by Roche to conduct the online survey part of the study. The authors declare that this study received funding from Roche Products Ltd/F. Hoffmann-La Roche. The funder (F. Hoffmann-La Roche) had the following involvement: qualitative interviews and survey parts of the study, design of the study and interpretation of the data. The funder (F. Hoffmann-La Roche) had the following involvement in the SUNFISH part 2 post-hoc analyses: design, analysis and interpretation of the data. Medical writing and editorial support were funded by F Hoffmann-La Roche., (Copyright © 2022 Duong, Staunton, Braid, Barriere, Trzaskoma, Gao, Willgoss, Cruz, Gusset, Gorni, Randhawa, Yang and Vuillerot.)

Published
2022
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47. Development of the SMA independence scale-upper limb module (SMAIS-ULM): A novel scale for individuals with Type 2 and non-ambulant Type 3 SMA.

Author

Trundell D, Skalicky A, Staunton H, Hareendran A, Le Scouiller S, Barrett L, Cooper O, Gorni K, Seabrook T, Jethwa S, and Cano S

Subjects
Cross-Sectional Studies, Humans, Psychometrics, Reproducibility of Results, Surveys and Questionnaires, Upper Extremity, Quality of Life, Spinal Muscular Atrophies of Childhood diagnosis
Abstract

Background: The amount of assistance required to perform daily activities for individuals with Type 2 and non-ambulant Type 3 spinal muscular atrophy (SMA) is often cited as meaningful for quality of life, and important to routinely assess., Methods: The SMA Independence Scale (SMAIS), a patient-reported outcome measure for individuals with SMA aged ≥12 years, and an observer-reported outcome measure for caregivers of individuals aged ≥2 years, was developed and evaluated in two phases. In Phase 1, 30 draft items were developed following review of the literature. Semi-structured interviews were then conducted with individuals with SMA and caregivers to establish content validity, resulting in a 29-item measure. In Phase 2, classical test theory and Rasch measurement theory methods were used to examine the cross-sectional and longitudinal measurement performance of the SMAIS in two independent datasets., Results: Phase 1 qualitative findings supported the relevance, acceptability, and comprehensibility of 29 items. In Phase 2, psychometric analyses indicated that the five response options were poorly discriminated and were thus collapsed to three options for subsequent analyses. Items showed statistical misfit, implying that the SMAIS was not assessing a single underlying construct. Based on conceptual evaluation of the items, and assessment of item performance, a more targeted 22-item upper limb score was derived. Reliability and validity analyses confirmed acceptable measurement properties of this score., Conclusions: Qualitative and quantitative analyses support the use of the 22-item SMAIS-Upper Limb Module in individuals with Type 2 and non-ambulant Type 3 SMA, aged ≥2 years., (Copyright © 2021 F Hoffmann-La Roche Ltd. Published by Elsevier B.V. All rights reserved.)

Published
2022
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48. Safety and efficacy of once-daily risdiplam in type 2 and non-ambulant type 3 spinal muscular atrophy (SUNFISH part 2): a phase 3, double-blind, randomised, placebo-controlled trial.

Author

Mercuri E, Deconinck N, Mazzone ES, Nascimento A, Oskoui M, Saito K, Vuillerot C, Baranello G, Boespflug-Tanguy O, Goemans N, Kirschner J, Kostera-Pruszczyk A, Servais L, Gerber M, Gorni K, Khwaja O, Kletzl H, Scalco RS, Staunton H, Yeung WY, Martin C, Fontoura P, and Day JW

Subjects
Adolescent, Adult, Aged, Azo Compounds adverse effects, Child, Child, Preschool, Double-Blind Method, Humans, Young Adult, Pyrimidines adverse effects, Spinal Muscular Atrophies of Childhood drug therapy, Spinal Muscular Atrophies of Childhood physiopathology
Abstract

Background: Risdiplam is an oral small molecule approved for the treatment of patients with spinal muscular atrophy, with approval for use in patients with type 2 and type 3 spinal muscular atrophy granted on the basis of unpublished data. The drug modifies pre-mRNA splicing of the SMN2 gene to increase production of functional SMN. We aimed to investigate the safety and efficacy of risdiplam in patients with type 2 or non-ambulant type 3 spinal muscular atrophy., Methods: In this phase 3, randomised, double-blind, placebo-controlled study, patients aged 2-25 years with confirmed 5q autosomal recessive type 2 or type 3 spinal muscular atrophy were recruited from 42 hospitals in 14 countries across Europe, North America, South America, and Asia. Participants were eligible if they were non-ambulant, could sit independently, and had a score of at least 2 in entry item A of the Revised Upper Limb Module. Patients were stratified by age and randomly assigned (2:1) to receive either daily oral risdiplam, at a dose of 5·00 mg (for individuals weighing ≥20 kg) or 0·25 mg/kg (for individuals weighing <20 kg), or daily oral placebo (matched to risdiplam in colour and taste). Randomisation was conducted by permutated block randomisation with a computerised system run by an external party. Patients, investigators, and all individuals in direct contact with patients were masked to treatment assignment. The primary endpoint was the change from baseline in the 32-item Motor Function Measure total score at month 12. All individuals who were randomly assigned to risdiplam or placebo, and who did not meet the prespecified missing item criteria for exclusion, were included in the primary efficacy analysis. Individuals who received at least one dose of risdiplam or placebo were included in the safety analysis. SUNFISH is registered with ClinicalTrials.gov, NCT02908685. Recruitment is closed; the study is ongoing., Findings: Between Oct 9, 2017, and Sept 4, 2018, 180 patients were randomly assigned to receive risdiplam (n=120) or placebo (n=60). For analysis of the primary endpoint, 115 patients from the risdiplam group and 59 patients from the placebo group were included. At month 12, the least squares mean change from baseline in 32-item Motor Function Measure was 1·36 (95% CI 0·61 to 2·11) in the risdiplam group and -0·19 (-1·22 to 0·84) in the placebo group, with a treatment difference of 1·55 (0·30 to 2·81, p=0·016) in favour of risdiplam. 120 patients who received risdiplam and 60 who received placebo were included in safety analyses. Adverse events that were reported in at least 5% more patients who received risdiplam than those who received placebo were pyrexia (25 [21%] of 120 patients who received risdiplam vs ten [17%] of 60 patients who received placebo), diarrhoea (20 [17%] vs five [8%]), rash (20 [17%] vs one [2%]), mouth and aphthous ulcers (eight [7%] vs 0), urinary tract infection (eight [7%] vs 0), and arthralgias (six [5%] vs 0). The incidence of serious adverse events was similar between treatment groups (24 [20%] of 120 patients in the risdiplam group; 11 [18%] of 60 patients in the placebo group), with the exception of pneumonia (nine [8%] in the risdiplam group; one [2%] in the placebo group)., Interpretation: Risdiplam resulted in a significant improvement in motor function compared with placebo in patients aged 2-25 years with type 2 or non-ambulant type 3 spinal muscular atrophy. Our exploratory subgroup analyses showed that motor function was generally improved in younger individuals and stabilised in older individuals, which requires confirmation in further studies. SUNFISH part 2 is ongoing and will provide additional evidence regarding the long-term safety and efficacy of risdiplam., Funding: F Hoffmann-La Roche., Competing Interests: Declaration of interests EM has received fees for serving on scientific advisory boards; speaker fees from F Hoffmann-La Roche, Biogen, AveXis/Novartis, Scholar Rock, and Cytokinetics; and grants from Biogen during the conduct of the study. ND served on scientific advisory boards for F Hoffmann-La Roche and Novartis pharmaceuticals; he has received personal fees from Biogen for congress and travel support. ESM reports that she has served on advisory boards for Biogen and Scholar Rock. She has received consulting fees, travel support, and speaker honoraria as an independent contractor from F Hoffmann-La Roche, Biogen, AveXis, and Scholar Rock. AN has received fees for serving on scientific advisory boards and speaker fees from F Hoffmann-La Roche. MO reports grants from F Hoffmann-La Roche during the conduct of the study and has received grants as a clinical trial investigator from Biogen. KS reports grants from F Hoffman-La Roche/Chugai Pharmaceutical during the conduct of the study and grants from Biogen Japan and lecture fees from Novartis Pharmaceuticals, outside the submitted work. CV reports personal fees and financial support to her institution from F Hoffmann-La Roche for activities outside the submitted work. GB received consultancy fees and speaker honoraria from F Hoffmann-La Roche and AveXis and speaker fees from PTC Therapeutics. NG reports fees for serving on advisory boards and presentations at symposia from F Hoffmann-La Roche, Biogen, and AveXis. JK received grants or contracts from Novartis Gene Therapies and Biogen. He has received consulting and speaker fees from F Hoffmann La Roche, Biogen, and Novartis Gene Therapies and consulting fees from Scholar Rock. AK-P reports that she received an institutional support grant from Biogen; serving on scientific advisory boards for and receiving speaker honoraria from Biogen, F Hoffmann-La Roche, Novartis, and PTC Therapeutics; and receiving personal fees from Biogen and F Hoffmann-La Roche for travel support. LS received grants and personal fees from F Hoffman-La Roche, Biogen, and AveXis/Novartis Gene Therapy and personal fees from Cytokinetics, outside the submitted work. OK reports that he was previously an employee and stockholder in F Hoffmann-La Roche during the submitted work and that he now holds the position of honorary chief medical officer at The Spinal Muscular Atrophy Foundation. JWD has received fees for serving on scientific advisory boards from Biogen, Novartis, Sarepta, and Avidity; has received consulting fees from Affinia Therapeutics and Shift Therapeutics for a therapeutic platform; and has received grant support for clinical trials from F Hoffman-La Roche, Biogen, Novartis Gene Therapies, Cytokinetics, Scholar Rock, and Sarepta. CM, PF, MG, KG, HK, HS, RSS, and WYY report that they are current employees and stockholders in F Hoffmann-La Roche. OB-T declared no competing interests., (Copyright © 2022 Elsevier Ltd. All rights reserved.)

Published
2022
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49. A Patient-Centered Conceptual Model of Symptoms and Their Impact in Early Parkinson's Disease: A Qualitative Study.

Author

Staunton H, Kelly K, Newton L, Leddin M, Rodriguez-Esteban R, Chaudhuri KR, Weintraub D, Postuma RB, and Martinez-Martin P

Subjects
Fatigue, Humans, Patient-Centered Care, Qualitative Research, Parkinson Disease complications, Parkinson Disease diagnosis, Quality of Life
Abstract

Background: Individuals with Parkinson's disease (PD) develop a significant disease burden over time that contributes to a progressive decline in health-related quality of life. There is a paucity of qualitative research to understand symptoms and impacts in individuals with early-stage PD (i.e., Hoehn and Yahr stage 1-2 and ≤2 years since diagnosis)., Objective: The collection of qualitative data to inform the selection of clinical outcome assessments for clinical trials is advocated by regulators. This patient-centered, multistage study sought to create a conceptual model of symptoms and their impact for individuals with early-stage PD., Methods: Symptoms and impacts of PD were gathered from a literature review of qualitative research, a quantitative social media listening analysis, and qualitative patient concept elicitation interviews (n = 35). Clinical experts provided input to validate and finalize the concepts., Results: The final conceptual model consisted of 27 symptoms categorized into 'motor' or 'non-motor' domains, and 39 impacts divided into five domains. Most frequently reported symptoms in early-stage PD were 'tremors' (89%), 'stiffness and rigidity', and 'fatigue' (69%, both). Most frequently reported impacts included 'anxiety' (74%), 'eating and drinking' (71%), followed by 'exercise/sport' and 'relationship with family/family life' (66%, both)., Conclusion: This study provides initial insights relating to the symptom and impact burden of early-stage PD patients. The conceptual model can be used to help researchers to develop and select optimal patient-centered outcomes to measure treatment benefit in clinical trials. These findings could inform future qualitative research and the development of outcomes specifically for early-stage PD patients.

Published
2022
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