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1. Assessing Self-Regulated Processes: What Do Primary School Students Do, Say and Think in the Process of Understanding a Text?

Author

Valentina Ronqui Leites, Daniel Trías Seferian, and Juan Antonio Huertas Martínez

Abstract

Self-regulation has a positive impact on learning and academic achievement, but due to its nature, it is difficult to assess it in a valid and reliable manner. This study aims to explore the validity of three self-regulation assessment methods in text comprehension tasks (questionnaire, think-aloud and traces) as well as to identify the variables associated with reading performance. For this purpose, individual sessions were conducted with 96 sixth-grade primary education students, who were asked to read a text and complete two reading comprehension tasks working in a virtual environment. Task traces were recorded, think-aloud was recorded and coded, and participants answered a self-report questionnaire about the strategies used. A limited relationship was found between self-regulation measured by means of the questionnaire, think-aloud and traces, and the relationship between think-aloud and traces was moderate. Regarding the factors related to text comprehension task performance, think-aloud and task time measurements were found to be better predictors of performance than the questionnaire.

Published
2024
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2. Comparing the Safety Action Feedback and Engagement (SAFE) Loop with an established incident reporting system: Study protocol for a pragmatic cluster randomized controlled trial.

Author

Berdahl, Carl, Henreid, Andrew, Cohen, Tara, Coleman, Bernice, Seferian, Edward, Leang, Donna, Kim, Sungjin, Diniz, Marcio, Grissinger, Matthew, Kaiser, Karen, McCleskey, Sara, Nuckols, Teryl, and Zhu, Xi

Subjects
Hospital incident reporting, Human factors engineering, Medical errors, Nursing care, Patient safety, Randomized controlled trial
Abstract

BACKGROUND: Incident reporting is widely used in hospitals to improve patient safety, but current reporting systems do not function optimally. The utility of incident reports is limited because hospital staff may not know what to report, may fear retaliation, and may doubt whether administrators will review reports and respond effectively. METHODS: This is a clustered randomized controlled trial of the Safety Action Feedback and Engagement (SAFE) Loop, an intervention designed to transform hospital incident reporting systems into effective tools for improving patient safety. The SAFE Loop has six key attributes: obtaining nurses input about which safety problems to prioritize on their unit; focusing on learning about selected high-priority events; training nurses to write more informative event reports; prompting nurses to report high-priority events; integrating information about events from multiple sources; and providing feedback to nurses on findings and mitigation plans. The study will focus on medication errors and randomize 20 nursing units at a large academic/community hospital in Los Angeles. Outcomes include: (1) incident reporting practices (rates of high-priority reports, contributing factors described in reports), (2) nurses attitudes toward incident reporting, and (3) rates of high-priority events. Quantitative analyses will compare changes in outcomes pre- and post-implementation between the intervention and control nursing units, and qualitative analyses will explore nurses experiences with implementation. CONCLUSION: If effective, SAFE Loop will have several benefits: increasing nurses engagement with reporting, producing more informative reports, enabling safety leaders to understand problems, designing system-based solutions more effectively, and lowering rates of high-priority patient safety events.

Published
2023

4. Respiration driven CO2 pulses dominate Australia's flux variability

Author

Metz, Eva-Marie, Vardag, Sanam N., Basu, Sourish, Jung, Martin, Ahrens, Bernhard, El-Madany, Tarek, Sitch, Stephen, Arora, Vivek K., Briggs, Peter R., Friedlingstein, Pierre, Goll, Daniel S., Jain, Atul K., Kato, Etsushi, Lombardozzi, Danica, Nabel, Julia E. M. S., Poulter, Benjamin, Séférian, Roland, Tian, Hanqin, Wiltshire, Andrew, Yuan, Wenping, Yue, Xu, Zaehle, Sönke, Deutscher, Nicholas M., Griffith, David W. T., and Butz, André

Subjects
Physics - Atmospheric and Oceanic Physics
Abstract

The Australian continent contributes substantially to the year-to-year variability of the global terrestrial carbon dioxide (CO2) sink. However, the scarcity of in-situ observations in remote areas prevents deciphering the processes that force the CO2 flux variability. Here, examining atmospheric CO2 measurements from satellites in the period 2009-2018, we find recurrent end-of-dry-season CO2 pulses over the Australian continent. These pulses largely control the year-to-year variability of Australia's CO2 balance, due to 2-3 times higher seasonal variations compared to previous top-down inversions and bottom-up estimates. The CO2 pulses occur shortly after the onset of rainfall and are driven by enhanced soil respiration preceding photosynthetic uptake in Australia's semi-arid regions. The suggested continental-scale relevance of soil rewetting processes has large implications for our understanding and modelling of global climate-carbon cycle feedbacks., Comment: 28 pages (including supplementary materials), 3 main figures, 7 supplementary figures; v2 changes: Last name of first author changed

Published
2022
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5. Motivación y autorregulación en el desempeño en matemáticas en estudiantes de Educación Secundaria

Author

Daniel Trías Seferian, Helena Sastre Abreu, and Olga Elena Cuadros-Jiménez

Subjects
aprendizaje, motivación, matemáticas, enseñanza secundaria, psicología de la educación, Education, Education (General), L7-991
Abstract

Contar con modelos que abran posibilidades de mejora de los desempeños en matemáticas constituye un desafío vigente para educadores e investigadores, particularmente en Latinoamérica. Este estudio se propone analizar las relaciones entre el desempeño en matemáticas, variables motivacionales, la autorregulación en el aprendizaje, la regulación emocional y el estatus socioeconómico. Se considera una muestra representativa que está constituida por 5344 adolescentes de tercer año de educación secundaria en Uruguay, procedente de datos secundarios obtenidos de la evaluación nacional de desempeño del sistema educativo uruguayo (ineed, 2020). Mediante análisis de ecuaciones estructurales se obtuvieron dos modelos con buenos niveles de ajuste, en los que el desempeño en matemáticas recoge incidencia directa del estatus socioeconómico y autoeficacia, mientras la motivación por la tarea, el sentido de pertenencia y la regulación emocional aportan fundamentalmente de modo indirecto; la autorregulación metacognitiva cumple un papel mediador, con baja incidencia directa. Los modelos recogen un conjunto de variables motivacionales y estrategias de autorregulación que son susceptibles de intervención educativa. Se discute sobre su consideración y abordaje en la enseñanza de las matemáticas en educación secundaria.

Published
2024
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8. Characteristics and outcomes of gemcitabine-associated pulmonary hypertension

Author

Pierre Mouillot, Nicolas Favrolt, Charles Khouri, Aurélie Grandvuillemin, Marie-Camille Chaumais, Déborah Schenesse, Andrei Seferian, Xavier Jais, Laurent Savale, Guillaume Beltramo, Olivier Sitbon, Jean-Luc Cracowski, Marc Humbert, Marjolaine Georges, Philippe Bonniaud, and David Montani

Subjects
Medicine
Abstract

Background Despite its known cardiac and lung toxicities, the chemotherapy drug gemcitabine has only rarely been associated with pulmonary hypertension (PH), and the underlying mechanism remains unclear. The objective of the present study was to assess the association between gemcitabine and PH. Methods We identified incident cases of precapillary PH confirmed by right heart catheterisation in patients treated with gemcitabine from the French PH Registry between January 2007 and December 2022. The aetiology, clinical, functional, radiological and haemodynamic characteristics of PH were reviewed at baseline and during follow-up. A pharmacovigilance disproportionality analysis was conducted using the World Health Organization (WHO) pharmacovigilance database. Results We identified nine cases of pulmonary arterial hypertension, either induced (in eight patients) or exacerbated (in one patient) by gemcitabine. Patients exhibited severe precapillary PH, with a median mean pulmonary arterial pressure of 40 (range 26–47) mmHg, a cardiac index of 2.4 (1.6–3.9) L·min−1·m−2 and a pulmonary vascular resistance of 6.3 (3.1–12.6) Wood units. The median time from the initiation of gemcitabine to the onset of PH was 7 (4–50) months, with patients receiving a median of 16 (6–24) gemcitabine injections. Six patients showed clinical improvement upon discontinuation of gemcitabine. In the WHO pharmacovigilance database, we identified a significant signal with 109 cases reporting at least one adverse event related to PH with gemcitabine. Conclusion Both clinical cases and pharmacovigilance data substantiate a significant association between gemcitabine use and the onset or worsening of precapillary PH. The observed improvement following the discontinuation of treatment underscores the importance of PH screening in gemcitabine-exposed patients experiencing unexplained dyspnoea.

Published
2024
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9. Safety, tolerability and pharmacokinetics of eteplirsen in young boys aged 6–48 months with Duchenne muscular dystrophy amenable to exon 51 skipping

Author

Deconinck, Nicolas, Van Coster, Rudy, Vanlander, Arnaud, Seferian, Andreea, De Lucia, Silvana, Gidaro, Teresa, Brande, Laura Vanden, Servais, Laurent, Kirschner, Janbernd, Borell, Sabine, Mercuri, Eugenio, Brogna, Claudia, Pane, Marika, Fanelli, Lavinia, Norcia, Giulia, Muntoni, Francesco, Brusa, Chiara, Chesshyre, Mary, Maresh, Kate, Pitchforth, Jaqueline, Schottlaender, Lucia, Scoto, Mariacristina, Silwal, Arpana, Trucco, Fedrica, Mercuri, E., Seferian, A.M., Servais, L., Deconinck, N., Stevenson, H., Ni, X., Zhang, W., East, L., Yonren, S., and Muntoni, F.

Published
2023
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12. Effects of gene replacement therapy with resamirigene bilparvovec (AT132) on skeletal muscle pathology in X-linked myotubular myopathy: results from a substudy of the ASPIRO open-label clinical trial

Author

Lawlor, Michael W., Schoser, Benedikt, Margeta, Marta, Sewry, Caroline A., Jones, Karra A., Shieh, Perry B., Kuntz, Nancy L., Smith, Barbara K., Dowling, James J., Müller-Felber, Wolfgang, Bönnemann, Carsten G., Seferian, Andreea M., Blaschek, Astrid, Neuhaus, Sarah, Foley, A. Reghan, Saade, Dimah N., Tsuchiya, Etsuko, Qasim, Ummulwara R., Beatka, Margaret, Prom, Mariah J., Ott, Emily, Danielson, Susan, Krakau, Paul, Kumar, Suresh N., Meng, Hui, Vanden Avond, Mark, Wells, Clive, Gordish-Dressman, Heather, Beggs, Alan H., Christensen, Sarah, Conner, Edward, James, Emma S., Lee, Jun, Sadhu, Chanchal, Miller, Weston, Sepulveda, Bryan, Varfaj, Fatbardha, Prasad, Suyash, and Rico, Salvador

Published
2024
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13. Outcomes and risk assessment in pulmonary veno-occlusive disease

Author

Athénaïs Boucly, Sabina Solinas, Antoine Beurnier, Xavier Jaïs, Sophia Keddache, Mélanie Eyries, Andrei Seferian, Mitja Jevnikar, Anne Roche, Sophie Bulifon, Arnaud Bourdin, Ari Chaouat, Vincent Cottin, Laurent Bertoletti, Laurent Savale, Marc Humbert, Olivier Sitbon, and David Montani

Subjects
Medicine
Abstract

Introduction Pulmonary veno-occlusive disease (PVOD) is a rare and severe subtype of pulmonary arterial hypertension (PAH). Although European Society of Cardiology/European Respiratory Society (ESC/ERS) guidelines advise assessing PAH severity at baseline and during follow-up, no existing risk assessment methods have been validated for PVOD. This study aimed to identify prognostic factors, examine the impact of treatment strategies and evaluate risk assessment methods for PVOD patients. Methods The study analysed all incident PVOD patients included in the French Pulmonary Hypertension Registry between 2006 and 2021. Survival was assessed based on initial treatment strategy and risk status and compared to a matched (age, sex, pulmonary vascular resistance) PAH group. Six risk assessment methods (number of four low-risk and three noninvasive low-risk variables, ESC/ERS guidelines three-strata and four-strata models, REVEAL 2.0 and Lite 2) were applied at baseline and early follow-up, and their accuracy was compared using Harrell's c-statistic. Results Among the 327 included PVOD patients, survival rates at 1, 3 and 5 years were 86%, 50% and 27%, respectively. Multivariate analysis showed that only 6-min walk distance was associated with survival, with no significant difference based on initial treatment strategy. All six risk assessment methods could discriminate mortality risk, and the ESC/ERS four-strata model was the most accurate at both baseline and follow-up (C-index 0.64 and 0.74). PVOD survival rates were consistently lower than PAH when comparing baseline risk status using the ESC/ERS four-strata model. Conclusion PVOD is associated with poor outcomes, and initial treatment strategies do not significantly affect survival. Risk assessment methods can be useful in predicting survival for PVOD patients.

Published
2024
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14. Safety and efficacy of gene replacement therapy for X-linked myotubular myopathy (ASPIRO): a multinational, open-label, dose-escalation trial

Author

Shieh, Perry B, Kuntz, Nancy L, Dowling, James J, Müller-Felber, Wolfgang, Bönnemann, Carsten G, Seferian, Andreea M, Servais, Laurent, Smith, Barbara K, Muntoni, Francesco, Blaschek, Astrid, Foley, A Reghan, Saade, Dimah N, Neuhaus, Sarah, Alfano, Lindsay N, Beggs, Alan H, Buj-Bello, Ana, Childers, Martin K, Duong, Tina, Graham, Robert J, Jain, Minal, Coats, Julie, MacBean, Vicky, James, Emma S, Lee, Jun, Mavilio, Fulvio, Miller, Weston, Varfaj, Fatbardha, Murtagh, Michael, Han, Cong, Noursalehi, Mojtaba, Lawlor, Michael W, Prasad, Suyash, and Rico, Salvador

Published
2023
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17. Comparing the Safety Action Feedback and Engagement (SAFE) Loop with an established incident reporting system: Study protocol for a pragmatic cluster randomized controlled trial

Author

Carl T. Berdahl, Andrew J. Henreid, Tara N. Cohen, Bernice L. Coleman, Edward G. Seferian, Donna Leang, Sungjin Kim, Marcio A. Diniz, Matthew Grissinger, Karen Kaiser, Sara McCleskey, Xi Zhu, and Teryl K. Nuckols

Subjects
Patient safety, Medical errors, Hospital incident reporting, Nursing care, Human factors engineering, Randomized controlled trial, Medicine (General), R5-920
Abstract

Background: Incident reporting is widely used in hospitals to improve patient safety, but current reporting systems do not function optimally. The utility of incident reports is limited because hospital staff may not know what to report, may fear retaliation, and may doubt whether administrators will review reports and respond effectively. Methods: This is a clustered randomized controlled trial of the Safety Action Feedback and Engagement (SAFE) Loop, an intervention designed to transform hospital incident reporting systems into effective tools for improving patient safety. The SAFE Loop has six key attributes: obtaining nurses' input about which safety problems to prioritize on their unit; focusing on learning about selected high-priority events; training nurses to write more informative event reports; prompting nurses to report high-priority events; integrating information about events from multiple sources; and providing feedback to nurses on findings and mitigation plans. The study will focus on medication errors and randomize 20 nursing units at a large academic/community hospital in Los Angeles. Outcomes include: (1) incident reporting practices (rates of high-priority reports, contributing factors described in reports), (2) nurses' attitudes toward incident reporting, and (3) rates of high-priority events. Quantitative analyses will compare changes in outcomes pre- and post-implementation between the intervention and control nursing units, and qualitative analyses will explore nurses’ experiences with implementation. Conclusion: If effective, SAFE Loop will have several benefits: increasing nurses’ engagement with reporting, producing more informative reports, enabling safety leaders to understand problems, designing system-based solutions more effectively, and lowering rates of high-priority patient safety events.

Published
2023
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18. Effect of weight-adjusted intermediate-dose versus fixed-dose prophylactic anticoagulation with low-molecular-weight heparin on venous thromboembolism among noncritically and critically ill patients with COVID-19: the COVI-DOSE trial, a multicenter, randomised, open-label, phase 4 trial

Author

Zuily, Stéphane, Couturaud, Francis, Goehringer, François, Laporte, Silvy, Mismetti, Patrick, Presles, Emilie, Rossignol, Patrick, Thilly, Nathalie, Wahl, Denis, Lecompte, Thomas, Oger, Emmanuel, Sevestre, Marie-Antoinette, Parent, Florence, Benhamou, Ygal, Roy, Pierre-Marie, Gigante, Tristan, Gilg, Morgane, Maclot, Pierre-Luc, Rossignol, Bénédicte, Udot, Jonathan, Bouteille, Hélène, Buchmuller, Andréa, Peurière, Céline, Siaghy, El Mehdi, Bouaziz, Valérie, Daguin, Charlotte, Grosjean, Benjamin, Saunier, Véronique, Verger, Laurence, Jacquot, Madlyne, Petitpain, Nadine, Charly, Martin, Tordella, Laurent, Baronnet, Guillaume, Baux, Elisabeth, Benetos, Athanase, Blanvillain, Jean-Baptiste, Conrad, Marie, Courte, Guilhem, Cravoisy-Popovic, Aurélie, Dufrost, Virginie, Gibot, Sébastien, Guerci, Philippe, Joly, Laure, Kimmoun, Antoine, Koszutski, Matthieu, Larue, Alexandrine, Levy, Bruno, Losser, Marie-Reine, Mattei, Mathieu, Nace, Lionel, Novy, Emmanuel, Perez, Pierre, Pertek, Jean-Pierre, Rigaux, Camille, Sauvage, Alexis, Schvoerer, Evelyne, Thivilier, Carine, Volkov, Lev, Zieminski, Piotr, Benlounes, Manil, Cheng, Charles, Fréminville, Jean-Baptiste de, Détriché, Grégoire, Flammarion, Emmanuel, Goudot, Guillaume, Hamdan, Amer, Hindré, Raphaël, Manoli, Corina, Messas, Emmanuel, Michon, Adrien, Mirault, Tristan, Pastré, Jean, Penet, Marie-Aude, Planquette, Benjamin, Volle, Geoffroy, Hamdan, Rémy, Laubriet-Jazayeri, Aline, Petit, Vincent, Piroth, Lionel, Quenot, Jean Pierre, Saccu, Mélissa, Barraud, Damien, Cavalli, Zoé, Gaci, Rostane, Andreu, Mathilde, Bertoletti, Laurent, De Magalhaes, Elodie, Bulifon, Sophie, Boucly, Athénaïs, Ebstein, Nathan, Humbert, Marc, Jaïs, Xavier, Jevnikar, Mitja, Savale, Laurent, Seferian, Andrei Horia, Colin, Charlotte, Ganem, Timothée, Roumila, Mehdi, Chopard, Romain, Besutti, Matthieu, Mouhat, Basile, Andrejak, Claire, Dupas, Stéphane, Le Roy, Gaëlle, Samy-Modeliar, Santhi, Coste, Anne, Fauche, Alexandre, Goetghebeur, David, Gut-Gobert, Christophe, Hoffmann, Clément, Hourmant, Baptiste, L'hévéder, Cécile, Lemoigne, Emmanuelle, Nepveu, Olivier, Paret, Raphaël, Picart, Gaël, Azerkan, Saïd, Boudaa, Chadia, Campagne, Julien, Eszto, Peter, Godbert, Benoît, Guichard, Jean-François, Heschung, Marion, Legoff, Antoine, Mariot, Jacques, Martin, Pascale, Mercy, Magalie, Perrin, Julie, Raymond, Stéphane, Vernier, Nathalie, Fesler, Pierre, Henneton, Pierrick, Mercuzot, Cédric, Pansu, Nathalie, Perez, Lucas, Andre, Loïc, Bakhache, Edgar, Chopin, Marie-Charlotte, Gilbert, Marie, Lambert, Marc, Pokeerbux, Mohammad Ryadh, Danion, François, Hansmann, Yves, Rougier, Estelle, Ruch, Yvon, Stéphan, Dominique, Ursenbach, Axel, Connerade, Isabelle, Gravier, Simon, Kayser, Damien, Michel, Jean-Marc, Mohseni, Mahsa, Younes, Waël, Burlacu, Ruxandra, Lopes, Amanda, Mouly, Stéphane, Nassarmadji, Kladoum, Sène, Damien, Siguret, Virginie, Stepanian, Alain, Annweiler, Cédric, Brangier, Antoine, Dubee, Vincent, Henni, Samir, Hersant, Jeanne, Loison, Jocelyne, Kern, Léa, Laine, Jean-Baptiste, Neveux-Brecheteau, Claire, Perez, Lucia, Benainous, Ruben, Giroux-Leprieur, Bénédicte, Lopez-Sublet, Marilucy, Khaled-Jousselin, Saïda, Bernard, Yohann, Amiot, Amélie, Breistroff, Jessica, Detry, Emilie, Diallo, Kadidiatou, Didier, Agnès, Dumont, Nathalie, Egensperger, Julie, Emmerich, Aurélie, François, Nelly, Gallo, Fanny, George, Valérie, Gérome, Quentin, Gutehrle, Aurélie, Lehman, Laure, Petit, Séverine, Piard, Vanessa, Saint-Gilles, Maximilien, Terenzi, Olivier, Marquette, Amélie, Mortelette, Hélène, Audry, Mathilde, Cransac, Amélie, Maillard, Marine, Boyer, Anaïs, Gallo, Floriana, Urbing, Arielle, Zahaf, Imane, Byczko, Alexandra, Chaalal, Amina, Berlier, Georgette, Bernabe, Corinne, Bezzeghoud, Souad, Chaudier, Caroline, Chauvet, Carole, Davier, Marina, Labruyere, Carine, Perrin, Estelle, Pierre, Michaël, Chekirine, Claire-Annissa, Voivret, Florence, Meftali, Ramdane, Sabri, Ouaffa, Beulaygue, Anaïs, Gall, Julie, Girard, Laure, Haddaoui, Soumia, Rami, Scheherazade, Couderc, Auriane, Le Breton, Aude, Perruche, Marie-Line, Claudon, Cindy, Roussel, Ludivine, Barnier, Aude, Blanchard, Tiphaine, Le Gall, Bénédicte, Pelouin, Mélanie, Veillon, Anne-Sophie, Aquereburu, Quam, Delaygue, Charlène, Ait Idir, Zahoua, Drugeon, Jérémy, Dubrulle, Déborah, Tezkratt, Rabah, Frantz, Anne-Sophie, Drouaine, Julie, Dubois, Jacqueline, Eyriey, Magali, Haerrel, Elina, Beaudenon, Mélinda, Guenet, Mialy, Lecerf, Thibaud, Marechal-Girault, Stéphanie, Rehaiem, Sami, Simon, Romain, Dangeul-Potier, Florence, Goulvent, Morgane, Fliss, Souha, Messani, Fadhila, Mizejewski, Béatrice, Mugnier, Brigitte, Opderbeck, Valérie, Risse, Brigitte, Lefèvre, Benjamin, Sanchez, Olivier, Empis de Vendin, Ombeline, de Ciancio, Guillaume, Arlet, Jean-Benoît, Khider, Lina, Terriat, Béatrice, Greigert, Hélène, Robert, Céline S., Louis, Guillaume, Trinh-Duc, Albert, Rispal, Patrick, Accassat, Sandrine, Thiery, Guillaume, Montani, David, Azarian, Réza, Meneveau, Nicolas, Soudet, Simon, Le Mao, Raphaël, Maurier, François, Le Moing, Vincent, Quéré, Isabelle, Yelnik, Cécile M., Lefebvre, Nicolas, Martinot, Martin, and Delrue, Maxime

Published
2023
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21. Safety and efficacy of risdiplam in patients with type 1 spinal muscular atrophy (FIREFISH part 2): secondary analyses from an open-label trial

Author

Volpe, Joseph J, Posner, John, Kellner, Ulrich, Quinlivan, Rosaline, Gerber, Marianne, Khwaja, Omar, Scalco, Renata S, Seabrook, Timothy, Koch, Armin, Balikova, Irina, Joniau, Inge, Accou, Geraldine, Tahon, Valentine, Wittevrongel, Sylvia, De Vos, Elke, de Holanda Mendonça, Rodrigo, Matsui Jr, Ciro, Fornazieri Darcie, Ana Letícia, Machado, Cleide, Kiyoko Oyamada, Maria, Martini, Joyce, Polido, Graziela, Rodrigues Iannicelli, Juliana, Caires de Oliveira Achili Ferreira, Juliana, Hu, Chaoping, Zhu, Xiaomei, Qian, Chen, Shen, Li, Li, Hui, Shi, Yiyun, Zhou, Shuizhen, Xiao, Ying, Zhou, Zhenxuan, Wang, Sujuan, Sang, Tian, Wei, Cuijie, Dong, Hui, Cao, Yiwen, Wen, Jing, Li, Wenzhu, Qin, Lun, Barisic, Nina, Celovec, Ivan, Galiot Delic, Martina, Ivkic, Petra Kristina, Vukojevic, Nenad, Kern, Ivana, Najdanovic, Boris, Skugor, Marin, Tomas, Josipa, Boespflug-Tanguy, Odile, De Lucia, Silvana, Seferian, Andrea, Barreau, Emmanuel, Mnafek, Nabila, Peche, Helene, Grange, Allison, Trang Nguyen, Diem, Milascevic, Darko, Tachibana, Shotaro, Pagliano, Emanuela, Bianchi Marzoli, Stefania, Santarsiero, Diletta, Garcia Sierra, Myriam, Tremolada, Gemma, Arnoldi, Maria Teresa, Vigano, Marta, Dosi, Claudia, Zanin, Riccardo, Schembri, Veronica, Brolatti, Noemi, Rao, Giuseppe, Tassara, Elisa, Morando, Simone, Tacchetti, Paola, Pedemonte, Marina, Priolo, Enrico, Sposetti, Lorenza, Comi, Giacomo Pietro, Govoni, Alessandra, Osnaghi, Silvia Gabriella, Minorini, Valeria, Abbati, Francesca, Fassini, Federica, Foa, Michaela, Lopopolo, Amalia, Pane, Marika, Palermo, Concetta, Pera, Maria Carmela, Amorelli, Giulia Maria, Barresi, Costanza, D'Amico, Guglielmo, Orazi, Lorenzo, Coratti, Giorgia, Leone, Daniela, Laura, Antonaci, De Sanctis, Roberto, Berti, Beatrice, Kimura, Naoki, Takeshima, Yasuhiro, Shimomura, Hideki, Lee, Tomoko, Gomi, Fumi, Morimatsu, Takanobu, Furukawa, Toru, Stodolska-Koberda, Urszula, Waskowska, Agnieszka, Kolendo, Jagoda, Sobierajska-Rek, Agnieszka, Modrzejewska, Sandra, Lemska, Anna, Melnik, Evgenia, Artemyeva, Svetlana, Leppenen, Natalya, Yupatova, Nataliya, Monakhova, Anastasya, Papina, Yulia, Shidlovsckaia, Olga, Litvinova, Elena, Enzmann, Cornelia, Galiart, Elea, Gugleta, Konstantin, Wondrusch Haschke, Christine, Topaloglu, Haluk, Oncel, Ibrahim, Ertugrul, Nesibe Eroglu, Konuskan, Bahadir, Eldem, Bora, Kadayifçilar, Sibel, Alemdaroglu, Ipek, Sari, Seher, Bilgin, Neslihan, Karaduman, Aynur Ayse, Sarikaya, Fatma Gokcem Yildiz, Graham, Robert J, Ghosh, Partha, Casavant, David, Levine, Alexis, Titus, Rachael, Engelbrekt, Amanda, Ambrosio, Lucia, Fulton, Anne, Baglieri, Anna Maria, Dias, Courtney, Maczek, Elizabeth, Pasternak, Amy, Beres, Shannon, Duong, Tina, Gee, Richard, Young, Sally, Masson, Riccardo, Mazurkiewicz-Bełdzińska, Maria, Rose, Kristy, Servais, Laurent, Xiong, Hui, Zanoteli, Edmar, Baranello, Giovanni, Bruno, Claudio, Day, John W, Deconinck, Nicolas, Klein, Andrea, Mercuri, Eugenio, Vlodavets, Dmitry, Wang, Yi, Dodman, Angela, El-Khairi, Muna, Gorni, Ksenija, Jaber, Birgit, Kletzl, Heidemarie, Gaki, Eleni, Fontoura, Paulo, and Darras, Basil T

Published
2022
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22. Functional respiratory complaints among COVID-19 survivors: a prospective cohort study

Author

Antoine Beurnier, Laurent Savale, Xavier Jaïs, Romain Colle, Tai Pham, Luc Morin, Sophie Bulifon, Nicolas Noël, Athénaïs Boucly, Benoit Delbarre, Nathan Ebstein, Samy Figueiredo, Matthieu Gasnier, Anatole Harrois, Etienne-Marie Jutant, Mitja Jevnikar, Sophia Keddache, Anne-Lise Lecoq, Olivier Meyrignac, Florence Parent, Jérémie Pichon, Mariana Preda, Anne Roche, Andrei Seferian, Marie-France Bellin, Thomas Gille, Emmanuelle Corruble, Olivier Sitbon, Laurent Becquemont, Xavier Monnet, Marc Humbert, David Montani, and and the Comebac Investigators

Subjects
Medicine
Abstract

Background Dyspnoea is a common persistent symptom after COVID-19. Whether it is associated with functional respiratory disorders remains unclear. Methods We assessed the proportion and characteristics of patients with “functional respiratory complaints” (FRCs) (as defined by Nijmegen Questionnaire >22) among 177 post-COVID-19 individuals who benefited from outclinic evaluation in the COMEBAC study (i.e., symptomatic and/or intensive care unit (ICU) survivors at 4 months). In a distinct explanatory cohort of 21 consecutive individuals with unexplained post-COVID-19 dyspnoea after routine tests, we also analysed the physiological responses to incremental cardiopulmonary exercise testing (CPET). Findings In the COMEBAC cohort, 37 patients had significant FRCs (20.9%, IC95: 14.9–26.9). The prevalence of FRCs ranged from 7.2% (ICU patients) to 37.5% (non-ICU patients). The presence of FRCs was significantly associated with more severe dyspnoea, lower 6-min walk distance, more frequent psychological and neurological symptoms (cognitive complaint, anxiety, depression, insomnia and post-traumatic stress disorders) and poorer quality of life (all p

Published
2023
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26. 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

Volpe, Joseph J., Posner, John, Kellner, Ulrich, Quinlivan, Rosaline, Daron, Aurore, Delstanche, Stéphanie, Bruninx, Romain, Dal Farra, Fabian, Schneider, Olivier, Deconinck, Nicolas, Balikova, Irina, Delbeke, Patricia, Joniau, Inge, Tahon, Valentine, Wittevrongel, Sylvia, De Vos, Elke, Goemans, Nathalie, Casteels, Ingele, De Waele, Liesbeth, Cassiman, Catherine, Prové, Lies, Kinoo, David, Vancampenhout, Lisa, Van Den Hauwe, Marleen, Van Impe, Annelies, Prufer de Queiroz Campos Araujo, Alexandra, Chacon Pereira, Aline, Nardes, Flávia, Haefeli, Lorena, Rossetto, Julia, Ferreira Rebel, Marcos, Almeida Pereira, Jaqueline, Campbell, Craig, Sharan, Sapna, McDonald, Wendy, Scholtes, Cheryl, Mah, Jean, Sframeli, Maria, Chiu, Angela, Hagel, Jane, Oskoui, Maryam, Beneish, Raquel, Cariou-Palmer, Gaela, Pham, Connie, Toffoli, Daniela, Arpin, Stephanie, Turgeon Desilets, Sarah, Wang, Yi, Hu, Chaoping, Huan, Jianfeng, Qian, Chen, Shen, Li, Xiao, Ying, Zhou, Zhenxuan, Li, Hui, Wang, Sujuan, Xiong, Hui, Chang, Xingzhi, Dong, Hui, Liu, Ying, Sang, Tian, Wei, Cuijie, Wen, Jing, Cao, Yiwen, Ly, Xingyao, Zhao, Jingjing, Li, Wenzhu, Qin, Lun, Barisic, Nina, Galiot Delic, Martina, Ivkic, Petra Kristina, Vukojevic, Nenad, Kern, Ivana, Najdanovic, Boris, Skugor, Marin, Servais, Laurent, Boespflug-Tanguy, Odile, Gidaro, Teresa, Seferian, Andreea, De Lucia, Silvana, Barreau, Emmanuel, Mnafek, Nabila, Momtchilova, Marta Milkova, Peche, Helene, Valherie, Carole, Grange, Allison, Lilien, Charlotte, Milascevic, Darko, Tachibana, Shotaro, Ravelli, Claudia, Cardas, Ruxandra, Taytard, Jessica, Aubertin, Guillaume, Vanden Brande, Laure, Davion, Jean-Baptiste, Coopman, Stephanie, Bouacha, Ikram, Debruyne, Philippe, Defoort, Sabine, Derlyn, Gilles, Leroy, Florian, Danjoux, Loïc, Guilbaud, Julie, Desguerre, Isabelle, Barnérias, Christine, Semeraro, Michaela, Bremond-Gignac, Dominique, Bruere, Lenaic, Rateaux, Maxence, Deladrière, Élodie, Germa, Virginie, Pereon, Yann, Mercie, Sandra, Billaud, Fanny, Le Goff, Lucie, Letellier, Guy, Vuillerot, Carole, Portefaix, Aurélie, De-Montferrand, Camille, Le-Goff, Laure, Fontaine, Stephanie, Saidi, Manel, Bouzid, Nabil, Barriere, Aurélie, Tinat, Marie, Kirschner, Janbernd, Dreesbach, Michelle, Lagréze, Wolf, Michaelis, Bettina, Molnar, Fanni, Seger, Dorina, Vogt, Sibylle, Bertini, Enrico, D'Amico, Adele, Petroni, Sergio, Bonetti, Anna Maria, Carlesi, Adelina, Mizzoni, Irene, Bruno, Claudio, Priolo, Enrico, Rao, Giuseppe, Morando, Simone, Tacchetti, Paola, Zuffi, Ambra, Comi, Giacomo Pietro, Brusa, Roberta, Corti, Stefania, Daniele, Velardo, Govoni, Alessandra, Magri, Francesca, Minorini, Valeria, Osnaghi, Silvia Gabriella, Abbati, Francesca, Fassini, Federica, Foa, Michaela, Lopopolo, Amaqlia, Meneri, Megi, Zoppas, Francesca, Parente, Valeria, Baranello, Giovanni, Masson, Riccardo, Bianchi Marzoli, Stefania, Santarsiero, Diletta, Garcia Sierra, Myriam, Tremolada, Gemma, Arnoldi, Maria Teresa, Vigano, Marta, Zanin, Riccardo, Mercuri, Eugenio, Antonaci, Laura, de Sanctis, Roberto, Pane, Marika, Pera, Maria Carmela, Amorelli, Giulia Maria, Barresi, Costanza, D'Amico, Gugliemo, Orazi, Lorenzo, Coratti, Giorgia, Haginoya, Kazuhiro, Kato, Atsuko, Morishita, Yuko, Kira, Ryutaro, Akiyama, Kiyomu, Goto, Miwako, Mori, Yujiro, Okamoto, Misato, Tsutsui, Saki, Takatsuji, Yuta, Tanaka, Aya, Komaki, Hirofumi, Omori, Miina, Suzuki, Ippei, Takeuchi, Mizuki, Todoroki, Daisuke, Watanabe, Seji, Matsubayashi, Tomoko, Inakazu, Emi, Nagura, Hiroe, Suzuki, Akira, Usui, Manami, Ishikawa, Nobutsune, Harada, Yousuke, Fudeyasu, Kenishi, Hirata, Kazuhiko, Michiue, Kana, Ueda, Kazuyuki, Saito, Kayoko, Fujitani, Junko, Arakawa, Reiko, Takano, Kozue, Yashiro, Shigeko, Seki, Maiko, Sano, Nozomi, Fukuyama, Koji, Matsumoto, Yuki, Miyazaki, Hirofumi, Shibata, Minoru, Kobayashi, Kyoko, Nakamura, Yukie, Takeshima, Yasuhiro, Kuma, Moe, Kostera-Pruszczyk, Anna, Fraczek, Anna, Jedrzejowska, Maria, Lusakowska, Anna, Czeszyk-Piotrowicz, Agnieszka, Hautz, Wojciech, Rakusiewicz, Klaudia, Burlewicz, Malgorzata, Gierlak-Wojcicka, Zuzanna, Kepa, Malwina, Sikorski, Adam, Sobieraj, Marcin, Mazurkiewicz-Beldzinska, Maria, Lemska, Anna, Modrzejewska, Sandra, Koberda, Mateusz, Stodolska-Koberda, Urszula, Waskowska, Agnieszka, Kolendo, Jagoda, Sobierajska-Rek, Agnieszka, Steinborn, Barbara, Dalz, Magdalena, Grabowska, Julia, Hajduk, Wojciech, Janasiewicz-Karachitos, Justyna, Klimas, Monika, Stopa, Marcin, Gajewska, Ewa, Pusz, Beata, Vlodavets, Dmitry, Melnik, Evgenia, Leppenen, Natalya, Yupatova, Nataliya, Monakhova, Anastasya, Papina, Yulia, Shidlovsckaia, Olga, Milic Rasic, Vedrana, Brankovic, Vesna, Kosac, Ana, Djokic, Olivera, Jakšic, Vesna, Pepic, Ana, Martinovic, Jelena, Munell Casadesus, Francina, Tizzano, Eduardo, Martín Begué, Nieves, Wolley Dod, Charlotte, Subira, Olaia, Planas Pascual, Bernat, Toro Tamargo, Esther, Madruga Garrido, Marcos, Medina Romero, José David, Salinas, Marta Peña, Nascimento Osorio, Andrés, Díaz Cortés, Ana, Jiménez Gañan, Enrique, Suh, Simone Dowon, Medina Cantillo, Julita, Moya, Obdulia, Padros, Nuria, Urraca, Sandra Roca, Valdivia, Hugo Gonzalez, Pascual Pascual, Samuel, de Manuel, Sofía, Martin, Susana Noval, Burnham, Paul, Espinosa, Sandra, Moreno, Mercedes Martinez, Topaloglu, Haluk, Oncel, Ibrahim, Eroglu Ertugru, Nesibe, Konuskan, Bahadir, Eldem, Bora, Kadayifçilar, Sibel, Alemdaroglu, Ipek, Karaduman, Aynur Ayse, Yilmaz, Oznur Tunca, Bilgin, Neslihan, Sari, Seher, Chiriboga, Claudia, Lee, John J., Rome-Martin, Donnielle, Day, John W., Beres, Shannon, Duong, Tina, Gee, Richard, Dunaway Young, Sally, Fuerst-Recktenwald, Sabine, Marquet, Anne, Muelhardt, Nicoletta, Trundell, Dylan, Mazzone, Elena S, Nascimento, Andres, Gerber, Marianne, Gorni, Ksenija, Khwaja, Omar, Kletzl, Heidemarie, Scalco, Renata S, Staunton, Hannah, Yeung, Wai Yin, Martin, Carmen, Fontoura, Paulo, and Day, John W

Published
2022
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27. European Joint Programme on Rare Diseases workshop: LAMA2-muscular dystrophy: paving the road to therapy March 17–19, 2023, Barcelona, Spain

Author

Smeets, Hubert, primary, Verbrugge, Bram, additional, Bulbena, Xavier, additional, Hristova, Liliya, additional, Vogt, Julia, additional, van Beckhoven, Isabelle, additional, Allamand, Valérie, additional, Almekinders, Josephine, additional, Barquinero, Jordi, additional, Berreur, Séverine, additional, Bönnemann, Carsten, additional, Bouman, Karlijn, additional, de Bruin, Laura, additional, Caron, Leslie, additional, Damon, Céline, additional, Durbeej, Madeleine, additional, Feijen, Doris, additional, Foley, Reghan, additional, Goncalves, Ana Rita, additional, Camelo, Clara Gontijo, additional, Güell, Marc, additional, Haliloğlu, Göknur, additional, Kemaladewi, Dwi, additional, Klein, Andrea, additional, Koleda, Nastia, additional, Minko, Olga, additional, Munell, Francina, additional, Nebermann, Tim, additional, Pini, Veronica, additional, Previtali, Stefano, additional, Roos, Andreas, additional, Rüegg, Markus, additional, Sarkozy, Anna, additional, Seferian, Andrea, additional, Smeets, Hubert, additional, Stepniewski, Jacek, additional, van Straten, Emma, additional, van Tienen, Florence, additional, Quijano-Roy, Susana, additional, Voermans, Nicol, additional, West, Alexia, additional, Yurchenco, Peter, additional, Moy, Justin, additional, Almeida, Cristina, additional, Becker, Johannes, additional, and Gill, Louise, additional

Published
2024
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28. Upper limb disease evolution in exon 53 skipping eligible patients with Duchenne muscular dystrophy

Author

Charlotte Lilien, Harmen Reyngoudt, Andreea Mihaela Seferian, Teresa Gidaro, Mélanie Annoussamy, Virginie Chê, Valérie Decostre, Isabelle Ledoux, Julien Le Louër, Eric Guemas, Francesco Muntoni, Jean‐Yves Hogrel, Pierre Georges Carlier, Laurent Servais, and and the PreU7 Study Group

Subjects
Neurosciences. Biological psychiatry. Neuropsychiatry, RC321-571, Neurology. Diseases of the nervous system, RC346-429
Abstract

Abstract Objective To understand the natural disease upper limb progression over 3 years of ambulatory and non‐ambulatory patients with Duchenne muscular dystrophy (DMD) using functional assessments and quantitative magnetic resonance imaging (MRI) and to exploratively identify prognostic factors. Methods Forty boys with DMD (22 non‐ambulatory and 18 ambulatory) with deletions in dystrophin that make them eligible for exon 53‐skipping therapy were included. Clinical assessments, including Brooke score, motor function measure (MFM), hand grip and key pinch strength, and upper limb distal coordination and endurance (MoviPlate), were performed every 6 months and quantitative MRI of fat fraction (FF) and lean muscle cross sectional area (flexor and extensor muscles) were performed yearly. Results In the whole population, there were strong nonlinear correlations between outcome measures. In non‐ambulatory patients, annual changes over the course of 3 years were detected with high sensitivity standard response mean (|SRM| ≥0.8) for quantitative MRI‐based FF, hand grip and key pinch, and MFM. Boys who presented with a FF27% were able to bring a glass to their mouth and retained this ability in the following 3 years. Ambulatory patients with grip strength >35% of predicted value and FF

Published
2021
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30. Onasemnogene abeparvovec gene therapy for symptomatic infantile-onset spinal muscular atrophy type 1 (STR1VE-EU): an open-label, single-arm, multicentre, phase 3 trial

Author

Seferian, A., De Lucia, S., Tachibana, S., Jollet, A., Mouffak, S., Pedemonte, M., Brolatti, N., Morando, S., Vanlander, A., De Vos, E., Tahon, V., Govoni, A., Magri, F., Comi, G., Foa, M., Parente, V., Buscemi, L., Dal Farra, F., Schneider, O., Jonas, A., Defeldre, A.C., Pagliano, E., Zanin, R., Arnoldi, M.T., Schembri, V., Del Sole, M., Mandelli, A., Pera, M.C., Antonaci, L., Coratti, G., de Sanctis, R., Pane, M., Scoto, M., Groves, K., Edel, L., Abel, F., Van Ruiten, H., Lofra, R.M., Thompson, E., Mercuri, Eugenio, Muntoni, Francesco, Baranello, Giovanni, Masson, Riccardo, Boespflug-Tanguy, Odile, Bruno, Claudio, Corti, Stefania, Daron, Aurore, Deconinck, Nicolas, Servais, Laurent, Straub, Volker, Ouyang, Haojun, Chand, Deepa, Tauscher-Wisniewski, Sitra, Mendonca, Nuno, and Lavrov, Arseniy

Published
2021
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32. A perspective on the next generation of Earth system model scenarios: towards representative emission pathways (REPs)

Author

Meinshausen, M, Schleussner, C-F, Beyer, K, Bodeker, G, Boucher, O, Canadell, JG, Daniel, JS, Diongue-Niang, A, Driouech, F, Fischer, E, Forster, P, Grose, M, Hansen, G, Hausfather, Z, Ilyina, T, Kikstra, JS, Kimutai, J, King, AD, Lee, J-Y, Lennard, C, Lissner, T, Nauels, A, Peters, GP, Pirani, A, Plattner, G-K, Poertner, H, Rogelj, J, Rojas, M, Roy, J, Samset, BH, Sanderson, BM, Seferian, R, Seneviratne, S, Smith, CJ, Szopa, S, Thomas, A, Urge-Vorsatz, D, Velders, GJM, Yokohata, T, Ziehn, T, Nicholls, Z, Meinshausen, M, Schleussner, C-F, Beyer, K, Bodeker, G, Boucher, O, Canadell, JG, Daniel, JS, Diongue-Niang, A, Driouech, F, Fischer, E, Forster, P, Grose, M, Hansen, G, Hausfather, Z, Ilyina, T, Kikstra, JS, Kimutai, J, King, AD, Lee, J-Y, Lennard, C, Lissner, T, Nauels, A, Peters, GP, Pirani, A, Plattner, G-K, Poertner, H, Rogelj, J, Rojas, M, Roy, J, Samset, BH, Sanderson, BM, Seferian, R, Seneviratne, S, Smith, CJ, Szopa, S, Thomas, A, Urge-Vorsatz, D, Velders, GJM, Yokohata, T, Ziehn, T, and Nicholls, Z

Abstract

In every Intergovernmental Panel on Climate Change (IPCC) Assessment cycle, a multitude of scenarios are assessed, with different scope and emphasis throughout the various Working Group reports and special reports, as well as their respective chapters. Within the reports, the ambition is to integrate knowledge on possible climate futures across the Working Groups and scientific research domains based on a small set of “framing pathways” such as the so-called representative concentration pathways (RCPs) in the Fifth IPCC Assessment Report (AR5) and the shared socioeconomic pathway (SSP) scenarios in the Sixth Assessment Report (AR6). This perspective, initiated by discussions at the IPCC Bangkok workshop in April 2023 on the “Use of Scenarios in AR6 and Subsequent Assessments”, is intended to serve as one of the community contributions to highlight the needs for the next generation of framing pathways that is being advanced under the Coupled Model Intercomparison Project (CMIP) umbrella, which will influence or even predicate the IPCC AR7 consideration of framing pathways. Here we suggest several policy research objectives that such a set of framing pathways should ideally fulfil, including mitigation needs for meeting the Paris Agreement objectives, the risks associated with carbon removal strategies, the consequences of delay in enacting that mitigation, guidance for adaptation needs, loss and damage, and for achieving mitigation in the wider context of societal development goals. Based on this context, we suggest that the next generation of climate scenarios for Earth system models should evolve towards representative emission pathways (REPs) and suggest key categories for such pathways. These framing pathways should address the most critical mitigation policy and adaptation plans that need to be implemented over the next 10 years. In our view, the most important categories are those relevant in the context of the Paris Agreement long-term goal, specific

Published
2024

35. Safety, tolerability and pharmacokinetics of eteplirsen in young boys aged 6–48 months with Duchenne muscular dystrophy amenable to exon 51 skipping

Author

E. Mercuri, A.M. Seferian, L. Servais, N. Deconinck, H. Stevenson, X. Ni, W. Zhang, L. East, S. Yonren, F. Muntoni, Nicolas Deconinck, Rudy Van Coster, Arnaud Vanlander, Andreea Seferian, Silvana De Lucia, Teresa Gidaro, Laura Vanden Brande, Laurent Servais, Janbernd Kirschner, Sabine Borell, Eugenio Mercuri, Claudia Brogna, Marika Pane, Lavinia Fanelli, Giulia Norcia, Francesco Muntoni, Chiara Brusa, Mary Chesshyre, Kate Maresh, Jaqueline Pitchforth, Lucia Schottlaender, Mariacristina Scoto, Arpana Silwal, and Fedrica Trucco

Subjects
Neurology, Pediatrics, Perinatology and Child Health, Neurology (clinical), Genetics (clinical)
Published
2023

36. Motivación y autorregulación en el desempeño en matemáticas en estudiantes de Educación Secundaria.

Author

Trías Seferian, Daniel, Sastre Abreu, Helena, and Elena Cuadros-Jiménez, Olga

Subjects
SELF-regulated learning, MATHEMATICS education, STRUCTURAL equation modeling, EMOTION regulation, MATHEMATICS education (Secondary)
Abstract

Copyright of Revista Colombiana de Educación is the property of Universidad Pedaggica Nacional and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract. (Copyright applies to all Abstracts.)

Published
2024
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37. Teaching to Self-Regulate in Mathematics: A Quasi-Experimental Study with Low-Achieving Elementary School Students

Author

Daniel Trias Seferian, Cindy Mels Auman, and Juan Antonio Huertas Martínez

Subjects
mathematics, metacognition, motivation, emotion, learning, Education
Abstract

Teaching students to self-regulate enhances their mathematics performance, yet few studies have investigated the long-term differential impact of particular self-regulation strategies specifically for low-achieving students. This quasi-experimental study evaluates the effect of teaching different self-regulation strategies on mathematical problem solving in low-achieving students. The participants were 69 sixth-grade elementary school students randomly assigned to one of three experimental groups (and taught predominantly cognitive, metacognitive or volitional strategies, while verifying intervention fidelity) or a control group for 16 sessions. Mathematical problem-solving skills were evaluated prior to the intervention, upon completion, and two months later. While all three intervention groups obtained significantly better results compared to the control group immediately after the intervention, volitional and metacognitive strategies showed the strongest and most lasting positive effects. We conclude that low-achieving students could benefit from learning self-regulation strategies, particularly when these strategies take into account the affective and motivational dynamics of learning.

Published
2021
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38. Natural history of Type 2 and 3 spinal muscular atrophy: 2‐year NatHis‐SMA study

Author

Mélanie Annoussamy, Andreea M. Seferian, Aurore Daron, Yann Péréon, Claude Cances, Carole Vuillerot, Liesbeth De Waele, Vincent Laugel, Ulrike Schara, Teresa Gidaro, Charlotte Lilien, Jean‐Yves Hogrel, Pierre Carlier, Emmanuel Fournier, Linda Lowes, Ksenija Gorni, Myriam Ly‐Le Moal, Nicole Hellbach, Timothy Seabrook, Christian Czech, Ricardo Hermosilla, Laurent Servais, and the NatHis‐SMA study group

Subjects
Neurosciences. Biological psychiatry. Neuropsychiatry, RC321-571, Neurology. Diseases of the nervous system, RC346-429
Abstract

Abstract Objective To characterize the natural history of spinal muscular atrophy (SMA) over 24 months using innovative measures such as wearable devices, and to provide evidence for the sensitivity of these measures to determine their suitability as endpoints in clinical trials. Methods Patients with Type 2 and 3 SMA (N = 81) with varied functional abilities (sitters, nonsitters, nonambulant, and ambulant) who were not receiving disease‐modifying treatment were assessed over 24 months: motor function (Motor Function Measure [MFM]), upper limb strength (MyoGrip, MyoPinch), upper limb activity (ActiMyo®), quantitative magnetic resonance imaging (fat fraction [FFT2] mapping and contractile cross‐sectional area [C‐CSA]), pulmonary function (forced vital capacity [FVC], peak cough flow, maximum expiratory pressure, maximum inspiratory pressure, and sniff nasal inspiratory pressure), and survival of motor neuron (SMN) protein levels. Results MFM32 scores declined significantly over 24 months, but not 12 months. Changes in upper limb activity could be detected over 6 months and continued to decrease significantly over 12 months, but not 24 months. Upper limb strength decreased significantly over 12 and 24 months. FVC declined significantly over 12 months, but not 24 months. FFT2 increased over 12 and 24 months, although not with statistical significance. A significant increase in C‐CSA was observed at 12 but not 24 months. Blood SMN protein levels were stable over 12 and 24 months. Interpretation These data demonstrate that the MFM32, MyoGrip, MyoPinch, and ActiMyo® enable the detection of a significant decline in patients with Type 2 and 3 SMA over 12 or 24 months.

Published
2021
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39. Hierarchical Bayesian modelling of disease progression to inform clinical trial design in centronuclear myopathy

Author

Eve Fouarge, Arnaud Monseur, Bruno Boulanger, Mélanie Annoussamy, Andreea M. Seferian, Silvana De Lucia, Charlotte Lilien, Leen Thielemans, Khazal Paradis, Belinda S. Cowling, Chris Freitag, Bradley P. Carlin, Laurent Servais, and the NatHis-MTM Study Group

Subjects
Bayesian analysis, Centronuclear myopathy, Complex innovative clinical trial design, Disease progression model, Natural history data, Medicine
Abstract

Abstract Background Centronuclear myopathies are severe rare congenital diseases. The clinical variability and genetic heterogeneity of these myopathies result in major challenges in clinical trial design. Alternative strategies to large placebo-controlled trials that have been used in other rare diseases (e.g., the use of surrogate markers or of historical controls) have limitations that Bayesian statistics may address. Here we present a Bayesian model that uses each patient’s own natural history study data to predict progression in the absence of treatment. This prospective multicentre natural history evaluated 4-year follow-up data from 59 patients carrying mutations in the MTM1 or DNM2 genes. Methods Our approach focused on evaluation of forced expiratory volume in 1 s (FEV1) in 6- to 18-year-old children. A patient was defined as a responder if an improvement was observed after treatment and the predictive probability of such improvement in absence of intervention was less than 0.01. An FEV1 response was considered clinically relevant if it corresponded to an increase of more than 8%. Results The key endpoint of a clinical trial using this model is the rate of response. The power of the study is based on the posterior probability that the rate of response observed is greater than the rate of response that would be observed in the absence of treatment predicted based on the individual patient’s previous natural history. In order to appropriately control for Type 1 error, the threshold probability by which the difference in response rates exceeds zero was adapted to 91%, ensuring a 5% overall Type 1 error rate for the trial. Conclusions Bayesian statistical analysis of natural history data allowed us to reliably simulate the evolution of symptoms for individual patients over time and to probabilistically compare these simulated trajectories to actual observed post-treatment outcomes. The proposed model adequately predicted the natural evolution of patients over the duration of the study and will facilitate a sufficiently powerful trial design that can cope with the disease’s rarity. Further research and ongoing dialog with regulatory authorities are needed to allow for more applications of Bayesian statistics in orphan disease research.

Published
2021
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40. Labor law contrarreforms and (surplus)valuation of collective bargaining in the context of sanitary crisis

Author

Gustavo Seferian

Subjects
negociação coletiva. precarização. mais-valorização. autonomia individual da vontade. covid-19., Law in general. Comparative and uniform law. Jurisprudence, K1-7720, International relations, JZ2-6530
Abstract

The article deals with the changes brought in the brazilian labor legislation granting collective bargaining permissiveness to make legal labor rights more flexible. Taking the historical and dialectical materialism as method, and discussing dogmatic, historical and social aspects of legality, the study seeks to demonstrate that this legislative change, despite having ideological traits that signal in a different sense, is a reflection of the bourgeois offensive against labor rights, evidencing that (i) the “valorization” of the collective composition does not bring prestige to social concertation as an effective mean for adjusting labor relations; (ii) that its main functionality is the induction to deepen the workforce exploitation, hence it can be read as an surplus-valuation of collective bargaining; (iii) serving as a relevant fissure to provide openings to the radicalization of the individual autonomy of the will, such as the judgment by the Brazilian Supreme Court of the ADI 6.363, in April 2020, in the context of the sanitary crisis resulting from the pandemic of COVID-19.

Published
2020
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41. Respiratory symptoms and radiological findings in post-acute COVID-19 syndrome

Author

Etienne-Marie Jutant, Olivier Meyrignac, Antoine Beurnier, Xavier Jaïs, Tai Pham, Luc Morin, Athénaïs Boucly, Sophie Bulifon, Samy Figueiredo, Anatole Harrois, Mitja Jevnikar, Nicolas Noël, Jérémie Pichon, Anne Roche, Andrei Seferian, Samer Soliman, Jacques Duranteau, Laurent Becquemont, Xavier Monnet, Olivier Sitbon, Marie-France Bellin, Marc Humbert, Laurent Savale, and David Montani

Subjects
Medicine
Abstract

Rationale The characteristics of patients with respiratory complaints and/or lung radiologic abnormalities after hospitalisation for coronavirus disease 2019 (COVID-19) are unknown. The objectives were to determine their characteristics and the relationships between dyspnoea, radiologic abnormalities and functional impairment. Methods In the COMEBAC (Consultation Multi-Expertise de Bicêtre Après COVID-19) cohort study, 478 hospital survivors were evaluated by telephone 4 months after hospital discharge, and 177 who had been hospitalised in an intensive care unit (ICU) or presented relevant symptoms underwent an ambulatory evaluation. New-onset dyspnoea and cough were evaluated, and the results of pulmonary function tests and high-resolution computed tomography of the chest were collected. Results Among the 478 patients, 78 (16.3%) reported new-onset dyspnoea, and 23 (4.8%) new-onset cough. The patients with new-onset dyspnoea were younger (56.1±12.3 versus 61.9±16.6 years), had more severe COVID-19 (ICU admission 56.4% versus 24.5%) and more frequent pulmonary embolism (18.0% versus 6.8%) (all p≤0.001) than patients without dyspnoea. Among the patients reassessed at the ambulatory care visit, the prevalence of fibrotic lung lesions was 19.3%, with extent

Published
2022
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42. The identification and management of interstitial lung disease in systemic sclerosis: evidence-based European consensus statements

Author

Hoffmann-Vold, Anna-Maria, Maher, Toby M, Philpot, Edward E, Ashrafzadeh, Ali, Barake, Rafic, Barsotti, Simone, Bruni, Cosimo, Carducci, Paolo, Carreira, Patricia E, Castellví, Ivan, Del Galdo, Francesco, Distler, Jörg H W, Foeldvari, Ivan, Fraticelli, Paolo, George, Peter M, Griffiths, Bridget, Guillén-Del-Castillo, Alfredo, Hamid, Abdul Monem, Horváth, Rudolf, Hughes, Michael, Kreuter, Michael, Moazedi-Fuerst, Florentine, Olas, Jacek, Paul, Suman, Rotondo, Cinzia, Rubio-Rivas, Manuel, Seferian, Andrei, Tomčík, Michal, Uzunhan, Yurdagül, Walker, Ulrich A, Więsik-Szewczyk, Ewa, and Distler, Oliver

Published
2020
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43. Hypertension pulmonaire associée à la gemcitabine : caractéristiques et évolution

Author

Mouillot, P., primary, Khoury, C., additional, Favrolt, N., additional, Grandvuillemin, A., additional, Chaumais, M.-C., additional, Schenesse, D., additional, Seferian, A., additional, Jais, X., additional, Savale, L., additional, Beltramo, G., additional, Sitbon, O., additional, Cracowski, J.-L., additional, Humbert, M., additional, Georges, M., additional, Bonniaud, P., additional, and Montani, D., additional

Published
2024
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45. Pathogenic DPAGT1 variants in limb‐girdle congenital myasthenic syndrome (LG‐CMS) associated with tubular aggregates and ORAI1 hypoglycosylation

Author

Brande, Laura Vanden, primary, Bauché, Stéphanie, additional, Pérez‐Guàrdia, Laura, additional, Sternberg, Damien, additional, Seferian, Andreea M., additional, Malfatti, Edoardo, additional, Silva‐Rojas, Roberto, additional, Labasse, Clémence, additional, Chevessier, Frédéric, additional, Carlier, Pierre, additional, Eymard, Bruno, additional, Romero, Norma B., additional, Laporte, Jocelyn, additional, Servais, Laurent, additional, Gidaro, Teresa, additional, and Böhm, Johann, additional

Published
2023
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47. Colony stimulating factor-1 (CSF-1) signalling is predictive of response to immune checkpoint inhibitors in advanced non-small cell lung cancer

Author

Takam Kamga, Paul, primary, Mayenga, Marie, additional, Sebane, Louise, additional, Costantini, Adrien, additional, Julie, Catherine, additional, Capron, Claude, additional, Parent, Florence, additional, Seferian, Andrei, additional, Guettier, Catherine, additional, Emile, Jean-François, additional, and Giroux Leprieur, Etienne, additional

Published
2023
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49. Decadal trends in the ocean carbon sink

Author

DeVries, Tim, LeQuéré, Corinne, Andrews, Oliver, Berthet, Sarah, Hauck, Judith, Ilyina, Tatiana, Landschützer, Peter, Lenton, Andrew, Lima, Ivan D., Nowicki, Michael, Schwinger, Jörg, and Séférian, Roland

Published
2019

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