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219 results on '"Bonanno, Silvia"'

1. Physical activity practiced at a young age is associated with a less severe subsequent clinical presentation in facioscapulohumeral muscular dystrophy

Author

Bettio, Cinzia, Banchelli, Federico, Salsi, Valentina, Vicini, Roberto, Crisafulli, Oscar, Ruggiero, Lucia, Ricci, Giulia, Bucci, Elisabetta, Angelini, Corrado, Berardinelli, Angela, Bonanno, Silvia, D’Angelo, Maria Grazia, Di Muzio, Antonio, Filosto, Massimiliano, Frezza, Erica, Maggi, Lorenzo, Mongini, Tiziana, Pegoraro, Elena, Rodolico, Carmelo, Scarlato, Marina, Vattemi, Gaetano, Velardo, Daniele, Tomelleri, Giuliano, D’Amico, Roberto, D’Antona, Giuseppe, and Tupler, Rossella

Published
2024
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3. Digital health and Clinical Patient Management System (CPMS) platform utility for data sharing of neuromuscular patients: the Italian EURO-NMD experience

Author

Fortunato, Fernanda, Bianchi, Francesca, Ricci, Giulia, Torri, Francesca, Gualandi, Francesca, Neri, Marcella, Farnè, Marianna, Giannini, Fabio, Malandrini, Alessandro, Volpi, Nila, Lopergolo, Diego, Silani, Vincenzo, Ticozzi, Nicola, Verde, Federico, Pareyson, Davide, Fenu, Silvia, Bonanno, Silvia, Nigro, Vincenzo, Peduto, Cristina, D’Ambrosio, Paola, Zeuli, Roberta, Zanobio, Mariateresa, Picillo, Esther, Servidei, Serenella, Primiano, Guido, Sancricca, Cristina, Sciacco, Monica, Brusa, Roberta, Filosto, Massimiliano, Cotti Piccinelli, Stefano, Pegoraro, Elena, Mongini, Tiziana, Solero, Luca, Gadaleta, Giulio, Brusa, Chiara, Minetti, Carlo, Bruno, Claudio, Panicucci, Chiara, Sansone, Valeria A., Lunetta, Christian, Zanolini, Alice, Toscano, Antonio, Pugliese, Alessia, Nicocia, Giulia, Bertini, Enrico, Catteruccia, Michela, Diodato, Daria, Atalaia, Antonio, Evangelista, Teresinha, Siciliano, Gabriele, and Ferlini, Alessandra

Published
2023
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5. Respiratory function in a large cohort of treatment-naïve adult spinal muscular atrophy patients: a cross-sectional study

Author

Vicino, Alex, Bello, Luca, Bonanno, Silvia, Govoni, Alessandra, Cerri, Federica, Ferraro, Manfredi, Capece, Giuliana, Gadaleta, Giulio, Meneri, Megi, Vacchiano, Veria, Ricci, Giulia, D'Errico, Eustachio, Tramacere, Irene, Banfi, Paolo, Bortolani, Sara, Zanin, Riccardo, Maioli, Maria Antonietta, Silvestrini, Mauro, Previtali, Stefano Carlo, Berardinelli, Angela, Turri, Mara, Coccia, Michela, Mantegazza, Renato, Liguori, Rocco, Filosto, Massimiliano, Siciliano, Gabriele, Simone, Isabella Laura, Mongini, Tiziana, Comi, Giacomo, Pegoraro, Elena, and Maggi, Lorenzo

Published
2023
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8. Six‐minute walk test as outcome measure of fatigability in adults with spinal muscular atrophy treated with nusinersen.

Author

Govoni, Alessandra, Ricci, Giulia, Bonanno, Silvia, Bello, Luca, Magri, Francesca, Meneri, Megi, Torri, Francesca, Caponnetto, Claudia, Passamano, Luigia, Grandis, Marina, Trojsi, Francesca, Cerri, Federica, Gadaleta, Giulio, Capece, Giuliana, Caumo, Luca, Tanel, Raffaella, Saccani, Elena, Vacchiano, Veria, Sorarù, Gianni, and D'Errico, Eustachio

Abstract

Introduction/Aims: Fatigue (subjective perception) and fatigability (objective motor performance worsening) are relevant aspects of disability in individuals with spinal muscular atrophy (SMA). The effect of nusinersen on fatigability in SMA patients has been investigated with conflicting results. We aimed to evaluate this in adult with SMA3. Methods: We conducted a multicenter retrospective cohort study, including adult ambulant patients with SMA3, data available on 6‐minute walk test (6MWT) and Hammersmith Functional Motor Scale—Expanded (HFMSE) at baseline and at least at 6 months of treatment with nusinersen. We investigated fatigability, estimated as 10% or higher decrease in walked distance between the first and sixth minute of the 6MWT, at baseline and over the 14‐month follow‐up. Results: Forty‐eight patients (56% females) were included. The 6MWT improved after 6, 10, and 14 months of treatment (p < 0.05). Of the 27 patients who completed the entire follow‐up, 37% improved (6MWT distance increase ≥30 m), 48.2% remained stable, and 14.8% worsened (6MWT distance decline ≥30 m). Fatigability was found at baseline in 26/38 (68%) patients and confirmed at subsequent time points (p < 0.05) without any significant change over the treatment period. There was no correlation between fatigability and SMN2 copy number, sex, age at disease onset, age at baseline, nor with 6MWT total distance and baseline HFMSE score. Discussion: Fatigability was detected at baseline in approximately 2/3 of SMA3 walker patients, without any correlation with clinical features, included motor performance. No effect on fatigability was observed during the 14‐month treatment period with nusinersen. [ABSTRACT FROM AUTHOR]

Published
2024
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10. Safety, efficacy, and tolerability of efgartigimod in patients with generalised myasthenia gravis (ADAPT): a multicentre, randomised, placebo-controlled, phase 3 trial

Author

De Bleecker, Jan L., De Koning, Kathy, De Mey, Katrien, De Pue, Annelien, Mercelis, Rudolf, Wyckmans, Maren, Vinck, Caroline, Wagemaekers, Linda, Baets, Jonathan, Ng, Eduardo, Shabanpour, Jafar, Daniyal, Lubna, Mannan, Shabber, Katzberg, Hans D., Genge, Angela, Siddiqi, Zaeem, Junkerová, Jana, Horakova, Jana, Reguliova, Katerina, Tyblova, Michaela, Jurajdova, Ivana, Novakova, Iveta, Jakubikova, Michala, Pitha, Jiri, Vohanka, Stanislav, Havelkova, Katerina, Horak, Tomas, Bednarik, Josef, Horakova, Mageda, Meisel, Andreas, Remstedt, Dike, Heibutzki, Claudia, Kohler, Siegfried, Gerischer, Lea, Hoffman, Sarah, Stascheit, Frauke, Vissing, John, Zafirakos, Lizzie, Khatri, Kuldeep Kumar, Autzen, Anne, Godtfeldt Stemmerik, Mads Peter, Andersen, Henning, Attarian, Shahram, Salort-Campana, Emmanuelle, Delmont, Emilien, Grapperon, Aude-Marie, Kouton, Ludivine, Tsiskaridze, Alexander, Rózsa, Csilla, Jakab, Gedeonne Margo, Toth, Szilvia, Szabo, Gyorgyi, Bors, David, Szabo, Eniko, Campanella, Angela, Vanoli, Fiammetta, Frangiamore, Rita, Antozzi, Carlo, Bonanno, Silvia, Maggi, Lorenzo, Giossi, Riccardo, Saccà, Francesco, Marsili, Angela, Pane, Chiara, Puorro, Giorgia, Reia, Antonio, Antonini, Giovanni, Alfieri, Girolamo, Morino, Stefania, Garibaldi, Matteo, Fionda, Laura, Leonardi, Luca, Konno, Shingo, Uzawa, Akiyuki, Sakuma, Kaoru, Watanabe, Chiho, Ozawa, Yukiko, Yasuda, Manato, Onishi, Yosuke, Samukawa, Makoto, Tsuda, Tomoko, Suzuki, Yasushi, Ishida, Sayaka, Watanabe, Genya, Takahashi, Masanori, Nakamura, Hiroko, Sugano, Erina, Kubota, Tomoya, Imai, Tomihiro, Suzuki., Mari, Mori, Ayako, Yamamoto, Daisuke, Ikeda, Kazuna, Hisahara, Shin, Masuda, Masayuki, Takaki, Miki, Minemoto, Kanako, Ido, Nobuhiro, Naito, Makiko, Okubo, Yoshihiko, Sugimoto, Takamichi, Takematsu, Yuka, Kamei, Ayumi, Shimizu, Mihiro, Naito, Hiroyuki, Nomura, Eiichi, Van Heur, Marjolein, Peters, Anne-Marie, Tannemaat, Martijn, Ruiter, Annabel, Keene, Kevin, Halas, Marek, Szczudlik, Andrzej, Pinkosz, Marta, Frasinska, Monika, Zwolinska, Grazyna, Kostera-Pruszczyk, Anna, Golenia, Aleksandra, Szczudlik, Piotr, Szczechowski, Lech, Pasko, Aneta, Poverennova, Irina, Urtaeva, Lubov, Kuznetsova, Nadezhda, Romanova, Tatiana, Nadezhda, Malkova, Lapochka, Elena, Korobko, Denis, Vergunova, Ilona, Melnikova, Anna, Bulatova, Ekaterina, Antipenko, Elena, Basta, Ivana, Bozovic, Ivo, Lavrnic, Dragana, Stojanovic, Vidosava Rakocevic, Beydoun, Said, Akhter, Salma, Malekniazi, Ali, Darki, Leila, Pimentel, Norianne, Cannon, Victoria, Chopra, Manisha, Traub, Rebecca, Mozaffar, Tahseen, Hernandez, Isela, Turner, Ivonne, Habib, Ali, Goyal, Namita, Kak, Manisha, Velasquez, Erik, Lam, Lucy, Suresh, Niraja, Farias, Jerrica, Jones, Sarah, Wagoner, Mary, Eggleston, Debbie, Bertorini, Tulio, Benzel, Cindy, Henegar, Robert, Pillai, Rekha, Bharavaju-Sanka, Ratna, Paiz, Carolyn, Jackson, Carlayne, Ruzhansky, Katherine, Dimitrova, Diana, Visser, Amy, Chahin, Nizar, Levine, Todd, Lisak, Robert, Jia, Kelly, Mada, Flicia, Bernitsas, Evanthia, Pasnoor, Mamatha, Roath, Katherine, Colgan, Samantha, Currence, Melissa, Heim, Andrew, Barohn, Richard, Dimachkie, Mazen, Statland, Jeffrey, Jawdat, Omar, Jabari, Duaa, Farmakidis, Constantine, Gilchrist, James, Li, Yuebing, Caristo, Irys, Hastings, Debbie, Morren, John Anthony, Weiss, Michael, Muppidi, Srikanth, Nguyen, Tia, Welsh, Lesly, So, Yuen, Goyal, Neelam, Pulley, Michael, Bailey, Cathy, Quraishi, Zubair, Berger, Alan, Sahagian, Gregory, Camberos, Yasmin, Frishberg, Benjamin, Howard, James F, Jr, Bril, Vera, Vu, Tuan, Karam, Chafic, Peric, Stojan, Margania, Temur, Murai, Hiroyuki, Bilinska, Malgorzata, Shakarishvili, Roman, Smilowski, Marek, Guglietta, Antonio, Ulrichts, Peter, Vangeneugden, Tony, Utsugisawa, Kimiaki, Verschuuren, Jan, and Mantegazza, Renato

Published
2021
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11. Efgartigimod in generalized myasthenia gravis: A real‐life experience at a national reference center

Author

Frangiamore, Rita, primary, Rinaldi, Elena, additional, Vanoli, Fiammetta, additional, Andreetta, Francesca, additional, Ciusani, Emilio, additional, Bonanno, Silvia, additional, Maggi, Lorenzo, additional, Gallone, Annamaria, additional, Colasuonno, Anna, additional, Tramacere, Irene, additional, Cheli, Marta, additional, Pinna, Alessandro, additional, Mantegazza, Renato, additional, and Antozzi, Carlo, additional

Published
2024
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12. Long-term efficacy and safety of eculizumab in Japanese patients with generalized myasthenia gravis: A subgroup analysis of the REGAIN open-label extension study

Author

Mazia, Claudio Gabriel, Wilken, Miguel, Barroso, Fabio, Saba, Juliet, Rugiero, Marcelo, Bettini, Mariela, Chaves, Marcelo, Vidal, Gonzalo, Garcia, Alejandra Dalila, De Bleecker, Jan, Van den Abeele, Guy, de Koning, Kathy, De Mey, Katrien, Mercelis, Rudy, Mahieu, Délphine, Wagemaekers, Linda, Van Damme, Philip, Depreitere, Annelies, Schotte, Caroline, Smetcoren, Charlotte, Stevens, Olivier, Van Daele, Sien, Vandenbussche, Nicolas, Vanhee, Annelies, Verjans, Sarah, Vynckier, Jan, D'Hondt, Ann, Tilkin, Petra, Alves de Siqueira Carvalho, Alzira, Dias Brockhausen, Igor, Feder, David, Ambrosio, Daniel, César, Pamela, Melo, Ana Paula, Martins Ribeiro, Renata, Rocha, Rosana, Bezerra Rosa, Bruno, Veiga, Thabata, da Silva, Luiz Augusto, Santos Engel, Murilo, Gonçalves Geraldo, Jordana, Ananias Morita, Maria da Penha, Nogueira Coelho, Erica, Paiva, Gabriel, Pozo, Marina, Prando, Natalia, Martineli Torres, Debora Dada, Butinhao, Cristiani Fernanda, Duran, Gustavo, Gomes da Silva, Tamires Cristina, Otavio Maia Gonçalves, Luiz, Pazetto, Lucas Eduardo, Fialho, Tomás Augusto Suriane, Renata Cubas Volpe, Luciana, Souza Duca, Luciana, Souza Bulle Oliveira, Acary, Amaral Andrade, Ana Carolina, Annes, Marcelo, Duarte Silva, Liene, Cavalcante Lino, Valeria, Pinto, Wladimir, Assis, Natália, Carrara, Fernanda, Miranda, Carolina, Souza, Iandra, Fernandes, Patricia, Siddiqi, Zaeem, Phan, Cecile, Narayan, Jeffrey, Blackmore, Derrick, Mallon, Ashley, Roderus, Rikki, Watt, Elizabeth, Junkerova, Jana, Kurkova, Barbora, Reguliova, Katarina, Zapletalova, Olga, Pitha, Jiri, Novakova, Iveta, Tyblova, Michaela, Jurajdova, Ivana, Wolfova, Marcela, Andersen, Henning, Harbo, Thomas, Vinge, Lotte, Krogh, Susanne, Mogensen, Anita, Vissing, John, Højgaard, Joan, Witting, Nanna, Mette Ostergaard Autzen, Anne, Pedersen, Jane, Eralinna, Juha-Pekka, Laaksonen, Mikko, Oksaranta, Olli, Harrison, Tuula, Eriksson, Jaana, Rozsa, Csilla, Horvath, Melinda, Lovas, Gabor, Matolcsi, Judit, Szabo, Gyorgyi, Jakab, Gedeonne, Szabadosne, Brigitta, Antonini, Giovanni, Di Pasquale, Antonella, Garibaldi, Matteo, Morino, Stefania, Troili, Fernanda, Fionda, Laura, Filla, Allessandro, Costabile, Teresa, Marano, Enrico, Saccà, Francesco, Fasanaro, Angiola, Marsili, Angela, Puorro, Giorgia, Mantegazza, Renato, Antozzi, Carlo, Bonanno, Silvia, Camera, Giorgia, Locatelli, Alberta, Maggi, Lorenzo, Pasanisi, Maria, Campanella, Angela, Evoli, Amelia, Alboini, Paolo Emilio, D'Amato, Valentina, Iorio, Raffaele, Kanai, Tetsuya, Kawaguchi, Naoki, Mori, Masahiro, Kaneko, Yoko, Kanzaki, Akiko, Kobayashi, Eri, Masaki, Katsuhisa, Matsuse, Dai, Matsushita, Takuya, Uehara, Taira, Shimpo, Misa, Jingu, Maki, Kikutake, Keiko, Nakamura, Yumiko, Sano, Yoshiko, Nagane, Yuriko, Kamegamori, Ikuko, Tsuda, Tomoko, Fujii, Yuko, Futono, Kazumi, Ozawa, Yukiko, Mizugami, Aya, Saito, Yuka, Morikawa, Miyuki, Samukawa, Makoto, Kamakura, Sachiko, Miyawaki, Eriko, Mitazaki, Teiichiro, Motomura, Masakatsu, Mukaino, Akihiro, Yoshimura, Shunsuke, Asada, Shizuka, Yoshida, Seiko, Amamoto, Shoko, Kobashikawa, Tomomi, Koga, Megumi, Maeda, Yasuko, Takada, Kazumi, Takada, Mihoko, Tsurumaru, Masako, Yamashita, Yumi, Akiyama, Tetsuya, Narikawa, Koichi, Tano, Ohito, Tsukita, Kenichi, Kurihara, Rikako, Meguro, Fumie, Fukuda, Yusuke, Sato, Miwako, Funaka, Soichiro, Kawamura, Tomohiro, Makamori, Masayuki, Takahashi, Masanori, Taichi, Namie, Hasuike, Tomoya, Higuchi, Eriko, Kobayashi, Hisako, Osakada, Kaori, Tsuda, Emiko, Shimohama, Shun, Hayashi, Takashi, Hisahara, Shin, Kawamata, Jun, Murahara, Takashi, Saitoh, Masaki, Suzuki, Shuichiro, Yamamoto, Daisuke, Ishiyama, Yoko, Ishiyama, Naoko, Noshiro, Mayuko, Takeyama, Rumi, Uwasa, Kaori, Yasuda, Ikuko, van der Kooi, Anneke, de Visser, Marianne, Gibson, Tamar, Casasnovas, Carlos, Alberti Aguilo, Maria Antonia, Homedes-Pedret, Christian, Julia Palacios, Natalia, Diez Porras, Laura, Velez Santamaria, Valentina, Lazaro, Ana, Diez Tejedor, Exuperio, Gomez Salcedo, Pilar, Fernandez-Fournier, Mireya, Lopez Ruiz, Pedro, Rodriguez de Rivera, Francisco Javier, Sastre, Maria, Gamez, Josep, Sune, Pilar, Salvado, Maria, Gili, Gisela, Mazuela, Gonzalo, Illa, Isabel, Cortes Vicente, Elena, Diaz-Manera, Jordi, Querol Gutierrez, Luis Antonio, Rojas Garcia, Ricardo, Vidal, Nuria, Arribas-Ibar, Elisabet, Piehl, Fredrik, Hietala, Albert, Bjarbo, Lena, Sengun, Ihsan, Meherremova, Arzu, Ozcelik, Pinar, Balkan, Bengu, Tuga, Celal, Ugur, Muzeyyen, Erdem-Ozdamar, Sevim, Bekircan-Kurt, Can Ebru, Acar, Nazire Pinar, Yilmaz, Ezgi, Caliskan, Yagmur, Orsel, Gulsah, Efendi, Husnu, Aydinlik, Seda, Cavus, Hakan, Kutlu, Ayse, Becerikli, Gulsah, Semiz, Cansu, Tun, Ozlem, Terzi, Murat, Dogan, Baki, Onar, Musa Kazim, Sen, Sedat, Kirbas Cavdar, Tugce, Veske, Adife, Norwood, Fiona, Dimitriou, Aikaterini, Gollogly, Jakit, Mahdi-Rogers, Mohamed, Seddigh, Arshira, Sokratous, Giannis, Maier, Gal, Sohail, Faisal, Jacob, Saiju, Sadalage, Girija, Torane, Pravin, Brown, Claire, Shah, Amna, Sathasivam, Sivakumar, Arndt, Heike, Davies, Debbie, Watling, Dave, Amato, Anthony, Cochrane, Thomas, Salajegheh, Mohammed, Roe, Kristen, Amato, Katherine, Toska, Shirli, Wolfe, Gil, Silvestri, Nicholas, Patrick, Kara, Zakalik, Karen, Katz, Jonathan, Miller, Robert, Engel, Marguerite, Forshew, Dallas, Bravver, Elena, Brooks, Benjamin, Plevka, Sarah, Burdette, Maryanne, Cunningham, Scott, Sanjak, Mohammad, Kramer, Megan, Nemeth, Joanne, Schommer, Clara, Tierney, Scott, Juel, Vern, Guptill, Jeffrey, Hobson-Webb, Lisa, Massey, Janice, Beck, Kate, Carnes, Donna, Loor, John, Anderson, Amanda, Pascuzzi, Robert, Bodkin, Cynthia, Kincaid, John, Snook, Riley, Guingrich, Sandra, Micheels, Angela, Chaudhry, Vinay, Corse, Andrea, Mosmiller, Betsy, Kelley, Andrea, Ho, Doreen, Srinivasan, Jayashri, Vytopil, Michal, Jara, Jordan, Ventura, Nicholas, Scala, Stephanie, Carter, Cynthia, Donahue, Craig, Herbert, Carol, Weiner, Elaine, Alam, Sharmeen, McKinnon, Jonathan, Haar, Laura, McKinnon, Naya, Alcon, Karan, McKenna, Kaitlyn, Sattar, Nadia, Daniels, Kevin, Jeffery, Dennis, Freimer, Miriam, Hoyle, Joseph Chad, Kissel, John, Agriesti, Julie, Chelnick, Sharon, Mezache, Louisa, Pineda, Colleen, Muharrem, Filiz, Karam, Chafic, Khoury, Julie, Marburger, Tessa, Kaur, Harpreet, Dimitrova, Diana, Gilchrist, James, Agrawal, Brajesh, Elsayed, Mona, Kohlrus, Stephanie, Andoin, Angela, Darnell, Taylor, Golden, Laura, Lokaitis, Barbara, Seelback, Jenna, Muppidi, Srikanth, Goyal, Neelam, Sakamuri, Sarada, So, Yuen T., Paulose, Shirley, Pol, Sabrina, Welsh, Lesly, Bhavaraju-Sanka, Ratna, Tobon Gonzales, Alejandro, Dishman, Lorraine, Jones, Floyd, Gonzalez, Anna, Padilla, Patricia, Saklad, Amy, Silva, Marcela, Kazamel, Mohamed, Alsharabati, Mohammad, Lu, Liang, Nozaki, Kenkichi, Mumfrey-Thomas, Sandi, Woodall, Amy, Mozaffar, Tahseen, Cash, Tiyonnoh, Goyal, Namita, Roy, Gulmohor, Mathew, Veena, Maqsood, Fatima, Minton, Brian, Jones, H. James, Rosenfeld, Jeffrey, Garcia, Rebekah, Echevarria, Laura, Garcia, Sonia, Pulley, Michael, Aranke, Shachie, Berger, Alan Ross, Shah, Jaimin, Shabbir, Yasmeen, Smith, Lisa, Varghese, Mary, Gutmann, Laurie, Gutmann, Ludwig, Jerath, Nivedita, Nance, Christopher, Swenson, Andrea, Olalde, Heena, Kressin, Nicole, Sieren, Jeri, Barohn, Richard, Dimachkie, Mazen, Glenn, Melanie, McVey, April, Pasnoor, Mamatha, Statland, Jeffery, Wang, Yunxia, Liu, Tina, Emmons, Kelley, Jenci, Nicole, Locheke, Jerry, Fondaw, Alex, Johns, Kathryn, Rico, Gabrielle, Walsh, Maureen, Herbelin, Laura, Hafer-Macko, Charlene, Kwan, Justin, Zilliox, Lindsay, Callison, Karen, Young, Valerie, DiSanzo, Beth, Naunton, Kerry, Benatar, Michael, Bilsker, Martin, Sharma, Khema, Cooley, Anne, Reyes, Eliana, Michon, Sara-Claude, Sheldon, Danielle, Steele, Julie, Howard Jr, James, Chopra, Manisha, Traub, Rebecca, Vu, Tuan, Katzin, Lara, McClain, Terry, Harvey, Brittany, Hart, Adam, Huynh, Kristin, Beydoun, Said, Chilingaryan, Amaiak, Doan, Victor, Droker, Brian, Gong, Hui, Karimi, Sanaz, Lin, Frank, Pokala, Krishna, Shah, Akshay, Tran, Anh, Akhter, Salma, Malekniazi, Ali, Tandan, Rup, Hehir, Michael, Waheed, Waqar, Lucy, Shannon, Weiss, Michael, Distad, Jane, Strom, Susan, Downing, Sharon, Kim, Bryan, Nowak, Richard, Dicapua, Daniel, Keung, Benison, Kumar, Aditya, Patwa, Huned, Robeson, Kimberly, Yang, Irene, Nye, Joan, Vu, Hong, Murai, Hiroyuki, Uzawa, Akiyuki, Suzuki, Yasushi, Imai, Tomihiro, Shiraishi, Hirokazu, Suzuki, Hidekazu, Okumura, Meinoshin, O’Brien, Fanny, Wang, Jing-Jing, Fujita, Kenji P., and Utsugisawa, Kimiaki

Published
2019
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13. Assessing the Role of Anti rh-GAA in Modulating Response to ERT in a Late-Onset Pompe Disease Cohort from the Italian GSDII Study Group

Author

Filosto, Massimiliano, Cotti Piccinelli, Stefano, Ravaglia, Sabrina, Servidei, Serenella, Moggio, Maurizio, Musumeci, Olimpia, Donati, Maria Alice, Pegoraro, Elena, Di Muzio, Antonio, Maggi, Lorenzo, Tonin, Paola, Marrosu, Gianni, Sancricca, Cristina, Lerario, Alberto, Sacchini, Michele, Semplicini, Claudio, Bozzoni, Virginia, Telese, Roberta, Bonanno, Silvia, Piras, Rachele, Maioli, Maria Antonietta, Ricci, Giulia, Vercelli, Liliana, Galvagni, Anna, Gallo Cassarino, Serena, Caria, Filomena, Mongini, Tiziana, Siciliano, Gabriele, Padovani, Alessandro, and Toscano, Antonio

Published
2019
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15. NGS-Based Genetic Analysis in a Cohort of Italian Patients with Suspected Inherited Myopathies and/or HyperCKemia

Author

Invernizzi, Federica, primary, Izzo, Rossella, additional, Colangelo, Isabel, additional, Legati, Andrea, additional, Zanetti, Nadia, additional, Garavaglia, Barbara, additional, Lamantea, Eleonora, additional, Peverelli, Lorenzo, additional, Ardissone, Anna, additional, Moroni, Isabella, additional, Maggi, Lorenzo, additional, Bonanno, Silvia, additional, Fiori, Laura, additional, Velardo, Daniele, additional, Magri, Francesca, additional, Comi, Giacomo P., additional, Ronchi, Dario, additional, Ghezzi, Daniele, additional, and Lamperti, Costanza, additional

Published
2023
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16. A human pan-genomic analysis reconfigures the genetic and epigenetic make up of facioscapulohumeral muscular dystrophy

Author

Salsi, Valentina, primary, Chiara, Matteo, additional, Pini, Sara, additional, Kuś, Paweł, additional, Ruggiero, Lucia, additional, Bonanno, Silvia, additional, Rodolico, Carmelo, additional, Previtali, Stefano C., additional, D’Angelo, Maria Grazia, additional, Maggi, Lorenzo, additional, Lopergolo, Diego, additional, Kimmel, Marek, additional, Santorelli, Filippo M., additional, Pesole, Graziano, additional, and Tupler, Rossella G., additional

Published
2023
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20. MiR-146a in ALS: Contribution to Early Peripheral Nerve Degeneration and Relevance as Disease Biomarker

Author

Giagnorio, Eleonora, primary, Malacarne, Claudia, additional, Cavalcante, Paola, additional, Scandiffio, Letizia, additional, Cattaneo, Marco, additional, Pensato, Viviana, additional, Gellera, Cinzia, additional, Riva, Nilo, additional, Quattrini, Angelo, additional, Dalla Bella, Eleonora, additional, Lauria, Giuseppe, additional, Mantegazza, Renato, additional, Bonanno, Silvia, additional, and Marcuzzo, Stefania, additional

Published
2023
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22. Adults with spinal muscular atrophy: a large-scale natural history study shows gender effect on disease

Author

Maggi, Lorenzo, primary, Bello, Luca, additional, Bonanno, Silvia, additional, Govoni, Alessandra, additional, Caponnetto, Claudia, additional, Passamano, Luigia, additional, Grandis, Marina, additional, Trojsi, Francesca, additional, Cerri, Federica, additional, Gardani, Alice, additional, Ferraro, Manfredi, additional, Gadaleta, Giulio, additional, Zangaro, Vittoria, additional, Caumo, Luca, additional, Maioli, Mariantonietta, additional, Tanel, Raffaella, additional, Saccani, Elena, additional, Meneri, Megi, additional, Vacchiano, Veria, additional, Ricci, Giulia, additional, Sorarù, Gianni, additional, D'Errico, Eustachio, additional, Bortolani, Sara, additional, Pavesi, Giovanni, additional, Gellera, Cinzia, additional, Zanin, Riccardo, additional, Corti, Stefania, additional, Silvestrini, Mauro, additional, Politano, Luisa, additional, Schenone, Angelo, additional, Previtali, Stefano Carlo, additional, Berardinelli, Angela, additional, Turri, Mara, additional, Verriello, Lorenzo, additional, Coccia, Michela, additional, Mantegazza, Renato, additional, Liguori, Rocco, additional, Filosto, Massimiliano, additional, Marrosu, Gianni, additional, Tiziano, Francesco Danilo, additional, Siciliano, Gabriele, additional, Simone, Isabella Laura, additional, Mongini, Tiziana, additional, Comi, Giacomo, additional, and Pegoraro, Elena, additional

Published
2022
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25. Identification of a cytokine profile in serum and cerebrospinal fluid of pediatric and adult spinal muscular atrophy patients and its modulation upon nusinersen treatment

Author

Bonanno, Silvia, primary, Cavalcante, Paola, additional, Salvi, Erika, additional, Giagnorio, Eleonora, additional, Malacarne, Claudia, additional, Cattaneo, Marco, additional, Andreetta, Francesca, additional, Venerando, Anna, additional, Pensato, Viviana, additional, Gellera, Cinzia, additional, Zanin, Riccardo, additional, Arnoldi, Maria Teresa, additional, Dosi, Claudia, additional, Mantegazza, Renato, additional, Masson, Riccardo, additional, Maggi, Lorenzo, additional, and Marcuzzo, Stefania, additional

Published
2022
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29. Anti-Spike IgG in multiple sclerosis patients after BNT162b2 vaccine: An exploratory case-control study in Italy

Author

Giossi, Riccardo, primary, Consonni, Alessandra, additional, Torri Clerici, Valentina, additional, Zito, Antonio, additional, Rigoni, Eleonora, additional, Antozzi, Carlo, additional, Brambilla, Laura, additional, Crisafulli, Sebastiano Giuseppe, additional, Bellino, Antonella, additional, Frangiamore, Rita, additional, Bonanno, Silvia, additional, Vanoli, Fiammetta, additional, Ciusani, Emilio, additional, Corsini, Elena, additional, Andreetta, Francesca, additional, Baggi, Fulvio, additional, Tramacere, Irene, additional, Mantegazza, Renato, additional, Conte, Antonella, additional, Bergamaschi, Roberto, additional, and Confalonieri, Paolo, additional

Published
2022
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32. 6MWT as measure of motor function and endurance in SMA type 3 patients treated with nusinersen

Author

Govoni, Alessandra, Ricci, Giulia, Maggi, Lorenzo, Meneri, Megi, Bonanno, Silvia, Bello, Luca, Caponnetto, Claudia, Passamano, Luigia, Grandis, Marina, Trojsi, Francesca, Cerri, Federica, Gadaleta, Giulio, Bozzoni, Virginia, Caumo, Luca, Piras, Rachele, Tanel, Raffaella, Saccani, Elena, Vacchiano, Veria, Sorarù, Gianni, D'Errico, Eustachio, Tramacere, Irene, Bortolani, Sara, Pavesi, Giovanni, Riccardo, Zanin, Silvestrini, Mauro, Politano, Luisa, Schenone, Angelo, Previtali, Stefano, Berardinelli, Angela, Turri, Mara, Verriello, Lorenzo, Coccia, Michela, Mantegazza, Renato, Liguori, Rocco, Filosto, Massimiliano, Maioli, Mariantonietta, Marrosu, Gianni, Simone, Isabella, Mongini, Tiziana Enrica, Pegoraro, Elena, Corti, Stefania, Siciliano, Gabriele, and Comi, Giacomo Pietro

Published
2021
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33. Dysregulation of myomiRs as common pathogenic feature associated with muscle atrophy in ALS, SMA and SBMA: Evidence from animal models and human patients

Author

Malacarne, Claudia, Galbiati, Mariarita, Giagnorio, Eleonora, Cavalcante, Paola, Salerno, Franco, Andreetta, Francesca, Cagnoli, Cinzia, Taiana, Michela, Nizzardo, Monica, Corti, Stefania, Pensato, Viviana, Venerando, Anna, Gellera, Cinzia, Fenu, Silvia, Pareyson, Davide, Masson, Riccardo, Maggi, Lorenzo, Bella, Eleonora Dalla, Lauria, Giuseppe, Mantegazza, Renato, Poletti, Angelo, Bonanno, Silvia, and Marcuzzo, Stefania

Published
2021
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35. Safety, efficacy, and tolerability of efgartigimod in patients with generalised myasthenia gravis (ADAPT): a multicentre, randomised, placebo-controlled, phase 3 trial

Author

Howard, James F, primary, Bril, Vera, additional, Vu, Tuan, additional, Karam, Chafic, additional, Peric, Stojan, additional, Margania, Temur, additional, Murai, Hiroyuki, additional, Bilinska, Malgorzata, additional, Shakarishvili, Roman, additional, Smilowski, Marek, additional, Guglietta, Antonio, additional, Ulrichts, Peter, additional, Vangeneugden, Tony, additional, Utsugisawa, Kimiaki, additional, Verschuuren, Jan, additional, Mantegazza, Renato, additional, De Bleecker, Jan L., additional, De Koning, Kathy, additional, De Mey, Katrien, additional, De Pue, Annelien, additional, Mercelis, Rudolf, additional, Wyckmans, Maren, additional, Vinck, Caroline, additional, Wagemaekers, Linda, additional, Baets, Jonathan, additional, Ng, Eduardo, additional, Shabanpour, Jafar, additional, Daniyal, Lubna, additional, Mannan, Shabber, additional, Katzberg, Hans D., additional, Genge, Angela, additional, Siddiqi, Zaeem, additional, Junkerová, Jana, additional, Horakova, Jana, additional, Reguliova, Katerina, additional, Tyblova, Michaela, additional, Jurajdova, Ivana, additional, Novakova, Iveta, additional, Jakubikova, Michala, additional, Pitha, Jiri, additional, Vohanka, Stanislav, additional, Havelkova, Katerina, additional, Horak, Tomas, additional, Bednarik, Josef, additional, Horakova, Mageda, additional, Meisel, Andreas, additional, Remstedt, Dike, additional, Heibutzki, Claudia, additional, Kohler, Siegfried, additional, Gerischer, Lea, additional, Hoffman, Sarah, additional, Stascheit, Frauke, additional, Vissing, John, additional, Zafirakos, Lizzie, additional, Khatri, Kuldeep Kumar, additional, Autzen, Anne, additional, Godtfeldt Stemmerik, Mads Peter, additional, Andersen, Henning, additional, Attarian, Shahram, additional, Salort-Campana, Emmanuelle, additional, Delmont, Emilien, additional, Grapperon, Aude-Marie, additional, Kouton, Ludivine, additional, Tsiskaridze, Alexander, additional, Rózsa, Csilla, additional, Jakab, Gedeonne Margo, additional, Toth, Szilvia, additional, Szabo, Gyorgyi, additional, Bors, David, additional, Szabo, Eniko, additional, Campanella, Angela, additional, Vanoli, Fiammetta, additional, Frangiamore, Rita, additional, Antozzi, Carlo, additional, Bonanno, Silvia, additional, Maggi, Lorenzo, additional, Giossi, Riccardo, additional, Saccà, Francesco, additional, Marsili, Angela, additional, Pane, Chiara, additional, Puorro, Giorgia, additional, Reia, Antonio, additional, Antonini, Giovanni, additional, Alfieri, Girolamo, additional, Morino, Stefania, additional, Garibaldi, Matteo, additional, Fionda, Laura, additional, Leonardi, Luca, additional, Konno, Shingo, additional, Uzawa, Akiyuki, additional, Sakuma, Kaoru, additional, Watanabe, Chiho, additional, Ozawa, Yukiko, additional, Yasuda, Manato, additional, Onishi, Yosuke, additional, Samukawa, Makoto, additional, Tsuda, Tomoko, additional, Suzuki, Yasushi, additional, Ishida, Sayaka, additional, Watanabe, Genya, additional, Takahashi, Masanori, additional, Nakamura, Hiroko, additional, Sugano, Erina, additional, Kubota, Tomoya, additional, Imai, Tomihiro, additional, Suzuki., Mari, additional, Mori, Ayako, additional, Yamamoto, Daisuke, additional, Ikeda, Kazuna, additional, Hisahara, Shin, additional, Masuda, Masayuki, additional, Takaki, Miki, additional, Minemoto, Kanako, additional, Ido, Nobuhiro, additional, Naito, Makiko, additional, Okubo, Yoshihiko, additional, Sugimoto, Takamichi, additional, Takematsu, Yuka, additional, Kamei, Ayumi, additional, Shimizu, Mihiro, additional, Naito, Hiroyuki, additional, Nomura, Eiichi, additional, Van Heur, Marjolein, additional, Peters, Anne-Marie, additional, Tannemaat, Martijn, additional, Ruiter, Annabel, additional, Keene, Kevin, additional, Halas, Marek, additional, Szczudlik, Andrzej, additional, Pinkosz, Marta, additional, Frasinska, Monika, additional, Zwolinska, Grazyna, additional, Kostera-Pruszczyk, Anna, additional, Golenia, Aleksandra, additional, Szczudlik, Piotr, additional, Szczechowski, Lech, additional, Pasko, Aneta, additional, Poverennova, Irina, additional, Urtaeva, Lubov, additional, Kuznetsova, Nadezhda, additional, Romanova, Tatiana, additional, Nadezhda, Malkova, additional, Lapochka, Elena, additional, Korobko, Denis, additional, Vergunova, Ilona, additional, Melnikova, Anna, additional, Bulatova, Ekaterina, additional, Antipenko, Elena, additional, Basta, Ivana, additional, Bozovic, Ivo, additional, Lavrnic, Dragana, additional, Stojanovic, Vidosava Rakocevic, additional, Beydoun, Said, additional, Akhter, Salma, additional, Malekniazi, Ali, additional, Darki, Leila, additional, Pimentel, Norianne, additional, Cannon, Victoria, additional, Chopra, Manisha, additional, Traub, Rebecca, additional, Mozaffar, Tahseen, additional, Hernandez, Isela, additional, Turner, Ivonne, additional, Habib, Ali, additional, Goyal, Namita, additional, Kak, Manisha, additional, Velasquez, Erik, additional, Lam, Lucy, additional, Suresh, Niraja, additional, Farias, Jerrica, additional, Jones, Sarah, additional, Wagoner, Mary, additional, Eggleston, Debbie, additional, Bertorini, Tulio, additional, Benzel, Cindy, additional, Henegar, Robert, additional, Pillai, Rekha, additional, Bharavaju-Sanka, Ratna, additional, Paiz, Carolyn, additional, Jackson, Carlayne, additional, Ruzhansky, Katherine, additional, Dimitrova, Diana, additional, Visser, Amy, additional, Chahin, Nizar, additional, Levine, Todd, additional, Lisak, Robert, additional, Jia, Kelly, additional, Mada, Flicia, additional, Bernitsas, Evanthia, additional, Pasnoor, Mamatha, additional, Roath, Katherine, additional, Colgan, Samantha, additional, Currence, Melissa, additional, Heim, Andrew, additional, Barohn, Richard, additional, Dimachkie, Mazen, additional, Statland, Jeffrey, additional, Jawdat, Omar, additional, Jabari, Duaa, additional, Farmakidis, Constantine, additional, Gilchrist, James, additional, Li, Yuebing, additional, Caristo, Irys, additional, Hastings, Debbie, additional, Morren, John Anthony, additional, Weiss, Michael, additional, Muppidi, Srikanth, additional, Nguyen, Tia, additional, Welsh, Lesly, additional, So, Yuen, additional, Goyal, Neelam, additional, Pulley, Michael, additional, Bailey, Cathy, additional, Quraishi, Zubair, additional, Berger, Alan, additional, Sahagian, Gregory, additional, Camberos, Yasmin, additional, and Frishberg, Benjamin, additional

Published
2021
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38. LncRNAs Associated with Neuronal Development and Oncogenesis Are Deregulated in SOD1-G93A Murine Model of Amyotrophic Lateral Sclerosis

Author

Rey, F, Marcuzzo, S, Bonanno, S, Bordoni, M, Giallongo, T, Malacarne, C, Cereda, C, Zuccotti, G, Carelli, S, Rey, Federica, Marcuzzo, Stefania, Bonanno, Silvia, Bordoni, Matteo, Giallongo, Toniella, Malacarne, Claudia, Cereda, Cristina, Zuccotti, Gian Vincenzo, Carelli, Stephana, Rey, F, Marcuzzo, S, Bonanno, S, Bordoni, M, Giallongo, T, Malacarne, C, Cereda, C, Zuccotti, G, Carelli, S, Rey, Federica, Marcuzzo, Stefania, Bonanno, Silvia, Bordoni, Matteo, Giallongo, Toniella, Malacarne, Claudia, Cereda, Cristina, Zuccotti, Gian Vincenzo, and Carelli, Stephana

Abstract

Amyotrophic Lateral Sclerosis (ALS) is a devastating neurodegenerative disease caused in 10% of cases by inherited mutations considered “familial”. An ever-increasing amount of evidence is showing a fundamental role for RNA metabolism in ALS pathogenesis, and long non-coding RNAs (lncRNAs) appear to play a role in ALS development. Here, we aim to investigate the expression of a panel of lncRNAs (linc-Enc1, linc–Brn1a, linc–Brn1b, linc-p21, Hottip, Tug1, Eldrr, and Fendrr) which could be implicated in early phases of ALS. Via Real-Time PCR, we assessed their expression in a murine familial model of ALS (SOD1-G93A mouse) in brain and spinal cord areas of SOD1-G93A mice in comparison with that of B6.SJL control mice, in asymptomatic (week 8) and late-stage disease (week 18). We highlighted a specific area and pathogenetic-stage deregulation in each lncRNA, with linc-p21 being deregulated in all analyzed tissues. Moreover, we analyzed the expression of their human homologues in SH-SY5Y-SOD1-WT and SH-SY5Y-SOD1-G93A, observing a profound alteration in their expression. Interestingly, the lncRNAs expression in our ALS models often resulted opposite to that observed for the lncRNAs in cancer. These evidences suggest that lncRNAs could be novel disease-modifying agents, biomarkers, or pathways affected by ALS neurodegeneration.

Published
2021

39. Dysregulation of Muscle-Specific MicroRNAs as Common Pathogenic Feature Associated with Muscle Atrophy in ALS, SMA and SBMA: Evidence from Animal Models and Human Patients

Author

Malacarne, Claudia, primary, Galbiati, Mariarita, additional, Giagnorio, Eleonora, additional, Cavalcante, Paola, additional, Salerno, Franco, additional, Andreetta, Francesca, additional, Cagnoli, Cinza, additional, Taiana, Michela, additional, Nizzardo, Monica, additional, Corti, Stefania, additional, Pensato, Viviana, additional, Venerando, Anna, additional, Gellera, Cinzia, additional, Fenu, Silvia, additional, Pareyson, Davide, additional, Masson, Riccardo, additional, Maggi, Lorenzo, additional, Dalla Bella, Eleonora, additional, Lauria, Giuseppe, additional, Mantegazza, Renato, additional, Bernasconi, Pia, additional, Poletti, Angelo, additional, Bonanno, Silvia, additional, and Marcuzzo, Stefania, additional

Published
2021
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41. Favorable outcome of COVID-19 infection in a patient with congenital myasthenic syndrome

Author

Bonanno, Silvia and Maggi, Lorenzo

Subjects
Pediatrics, medicine.medical_specialty, Neurology, Coronavirus disease 2019 (COVID-19), Adolescent, Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2), Clinical Neurology, MEDLINE, Dermatology, Congenital myasthenic syndrome, Positive outcome, COVID-19 Testing, Medicine, Humans, Favorable outcome, Neuroradiology, Myasthenic Syndromes, Congenital, business.industry, COVID-19, General Medicine, medicine.disease, Psychiatry and Mental health, Treatment Outcome, Female, Neurology (clinical), Neurosurgery, business, Neuromuscular disorders
Abstract

Patients affected by neuromuscular disorders (NMDs) are theoretically at higher risk for severe illness from SARS-CoV2 due to respiratory and swallowing muscle weakness 1. Here we describe the first case of congenital myasthenic syndrome (CMS) who was infected with COVID-19 and showed a positive outcome.

Published
2020

42. CXCL13/CXCR5 signalling is pivotal to preserve motor neurons in amyotrophic lateral sclerosis

Author

Trolese, Maria Chiara, primary, Mariani, Alessandro, additional, Terao, Mineko, additional, de Paola, Massimiliano, additional, Fabbrizio, Paola, additional, Sironi, Francesca, additional, Kurosaki, Mami, additional, Bonanno, Silvia, additional, Marcuzzo, Stefania, additional, Bernasconi, Pia, additional, Trojsi, Francesca, additional, Aronica, Eleonora, additional, Bendotti, Caterina, additional, and Nardo, Giovanni, additional

Published
2020
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44. Nusinersen safety and effects on motor function in adult spinal muscular atrophy type 2 and 3

Author

Maggi, Lorenzo, primary, Bello, Luca, additional, Bonanno, Silvia, additional, Govoni, Alessandra, additional, Caponnetto, Claudia, additional, Passamano, Luigia, additional, Grandis, Marina, additional, Trojsi, Francesca, additional, Cerri, Federica, additional, Ferraro, Manfredi, additional, Bozzoni, Virginia, additional, Caumo, Luca, additional, Piras, Rachele, additional, Tanel, Raffaella, additional, Saccani, Elena, additional, Meneri, Megi, additional, Vacchiano, Veria, additional, Ricci, Giulia, additional, Soraru', Gianni, additional, D'Errico, Eustachio, additional, Tramacere, Irene, additional, Bortolani, Sara, additional, Pavesi, Giovanni, additional, Zanin, Riccardo, additional, Silvestrini, Mauro, additional, Politano, Luisa, additional, Schenone, Angelo, additional, Previtali, Stefano Carlo, additional, Berardinelli, Angela, additional, Turri, Mara, additional, Verriello, Lorenzo, additional, Coccia, Michela, additional, Mantegazza, Renato, additional, Liguori, Rocco, additional, Filosto, Massimiliano, additional, Marrosu, Gianni, additional, Siciliano, Gabriele, additional, Simone, Isabella Laura, additional, Mongini, Tiziana, additional, Comi, Giacomo, additional, and Pegoraro, Elena, additional

Published
2020
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46. Circulating MyomiRs as Potential Biomarkers to Monitor Response to Nusinersen in Pediatric SMA Patients

Author

Bonanno, Silvia, primary, Marcuzzo, Stefania, additional, Malacarne, Claudia, additional, Giagnorio, Eleonora, additional, Masson, Riccardo, additional, Zanin, Riccardo, additional, Arnoldi, Maria Teresa, additional, Andreetta, Francesca, additional, Simoncini, Ornella, additional, Venerando, Anna, additional, Gellera, Cinzia, additional, Pantaleoni, Chiara, additional, Mantegazza, Renato, additional, Bernasconi, Pia, additional, Baranello, Giovanni, additional, and Maggi, Lorenzo, additional

Published
2020
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48. Long-term efficacy and safety of eculizumab in Japanese patients with generalized myasthenia gravis: A subgroup analysis of the REGAIN open-label extension study

Author

Murai, Hiroyuki, primary, Uzawa, Akiyuki, additional, Suzuki, Yasushi, additional, Imai, Tomihiro, additional, Shiraishi, Hirokazu, additional, Suzuki, Hidekazu, additional, Okumura, Meinoshin, additional, O’Brien, Fanny, additional, Wang, Jing-Jing, additional, Fujita, Kenji P., additional, Utsugisawa, Kimiaki, additional, Mazia, Claudio Gabriel, additional, Wilken, Miguel, additional, Barroso, Fabio, additional, Saba, Juliet, additional, Rugiero, Marcelo, additional, Bettini, Mariela, additional, Chaves, Marcelo, additional, Vidal, Gonzalo, additional, Garcia, Alejandra Dalila, additional, De Bleecker, Jan, additional, Van den Abeele, Guy, additional, de Koning, Kathy, additional, De Mey, Katrien, additional, Mercelis, Rudy, additional, Mahieu, Délphine, additional, Wagemaekers, Linda, additional, Van Damme, Philip, additional, Depreitere, Annelies, additional, Schotte, Caroline, additional, Smetcoren, Charlotte, additional, Stevens, Olivier, additional, Van Daele, Sien, additional, Vandenbussche, Nicolas, additional, Vanhee, Annelies, additional, Verjans, Sarah, additional, Vynckier, Jan, additional, D'Hondt, Ann, additional, Tilkin, Petra, additional, Alves de Siqueira Carvalho, Alzira, additional, Dias Brockhausen, Igor, additional, Feder, David, additional, Ambrosio, Daniel, additional, César, Pamela, additional, Melo, Ana Paula, additional, Martins Ribeiro, Renata, additional, Rocha, Rosana, additional, Bezerra Rosa, Bruno, additional, Veiga, Thabata, additional, da Silva, Luiz Augusto, additional, Santos Engel, Murilo, additional, Gonçalves Geraldo, Jordana, additional, Ananias Morita, Maria da Penha, additional, Nogueira Coelho, Erica, additional, Paiva, Gabriel, additional, Pozo, Marina, additional, Prando, Natalia, additional, Martineli Torres, Debora Dada, additional, Butinhao, Cristiani Fernanda, additional, Duran, Gustavo, additional, Gomes da Silva, Tamires Cristina, additional, Otavio Maia Gonçalves, Luiz, additional, Pazetto, Lucas Eduardo, additional, Fialho, Tomás Augusto Suriane, additional, Renata Cubas Volpe, Luciana, additional, Souza Duca, Luciana, additional, Souza Bulle Oliveira, Acary, additional, Amaral Andrade, Ana Carolina, additional, Annes, Marcelo, additional, Duarte Silva, Liene, additional, Cavalcante Lino, Valeria, additional, Pinto, Wladimir, additional, Assis, Natália, additional, Carrara, Fernanda, additional, Miranda, Carolina, additional, Souza, Iandra, additional, Fernandes, Patricia, additional, Siddiqi, Zaeem, additional, Phan, Cecile, additional, Narayan, Jeffrey, additional, Blackmore, Derrick, additional, Mallon, Ashley, additional, Roderus, Rikki, additional, Watt, Elizabeth, additional, Junkerova, Jana, additional, Kurkova, Barbora, additional, Reguliova, Katarina, additional, Zapletalova, Olga, additional, Pitha, Jiri, additional, Novakova, Iveta, additional, Tyblova, Michaela, additional, Jurajdova, Ivana, additional, Wolfova, Marcela, additional, Andersen, Henning, additional, Harbo, Thomas, additional, Vinge, Lotte, additional, Krogh, Susanne, additional, Mogensen, Anita, additional, Vissing, John, additional, Højgaard, Joan, additional, Witting, Nanna, additional, Mette Ostergaard Autzen, Anne, additional, Pedersen, Jane, additional, Eralinna, Juha-Pekka, additional, Laaksonen, Mikko, additional, Oksaranta, Olli, additional, Harrison, Tuula, additional, Eriksson, Jaana, additional, Rozsa, Csilla, additional, Horvath, Melinda, additional, Lovas, Gabor, additional, Matolcsi, Judit, additional, Szabo, Gyorgyi, additional, Jakab, Gedeonne, additional, Szabadosne, Brigitta, additional, Antonini, Giovanni, additional, Di Pasquale, Antonella, additional, Garibaldi, Matteo, additional, Morino, Stefania, additional, Troili, Fernanda, additional, Fionda, Laura, additional, Filla, Allessandro, additional, Costabile, Teresa, additional, Marano, Enrico, additional, Saccà, Francesco, additional, Fasanaro, Angiola, additional, Marsili, Angela, additional, Puorro, Giorgia, additional, Mantegazza, Renato, additional, Antozzi, Carlo, additional, Bonanno, Silvia, additional, Camera, Giorgia, additional, Locatelli, Alberta, additional, Maggi, Lorenzo, additional, Pasanisi, Maria, additional, Campanella, Angela, additional, Evoli, Amelia, additional, Alboini, Paolo Emilio, additional, D'Amato, Valentina, additional, Iorio, Raffaele, additional, Kanai, Tetsuya, additional, Kawaguchi, Naoki, additional, Mori, Masahiro, additional, Kaneko, Yoko, additional, Kanzaki, Akiko, additional, Kobayashi, Eri, additional, Masaki, Katsuhisa, additional, Matsuse, Dai, additional, Matsushita, Takuya, additional, Uehara, Taira, additional, Shimpo, Misa, additional, Jingu, Maki, additional, Kikutake, Keiko, additional, Nakamura, Yumiko, additional, Sano, Yoshiko, additional, Nagane, Yuriko, additional, Kamegamori, Ikuko, additional, Tsuda, Tomoko, additional, Fujii, Yuko, additional, Futono, Kazumi, additional, Ozawa, Yukiko, additional, Mizugami, Aya, additional, Saito, Yuka, additional, Morikawa, Miyuki, additional, Samukawa, Makoto, additional, Kamakura, Sachiko, additional, Miyawaki, Eriko, additional, Mitazaki, Teiichiro, additional, Motomura, Masakatsu, additional, Mukaino, Akihiro, additional, Yoshimura, Shunsuke, additional, Asada, Shizuka, additional, Yoshida, Seiko, additional, Amamoto, Shoko, additional, Kobashikawa, Tomomi, additional, Koga, Megumi, additional, Maeda, Yasuko, additional, Takada, Kazumi, additional, Takada, Mihoko, additional, Tsurumaru, Masako, additional, Yamashita, Yumi, additional, Akiyama, Tetsuya, additional, Narikawa, Koichi, additional, Tano, Ohito, additional, Tsukita, Kenichi, additional, Kurihara, Rikako, additional, Meguro, Fumie, additional, Fukuda, Yusuke, additional, Sato, Miwako, additional, Funaka, Soichiro, additional, Kawamura, Tomohiro, additional, Makamori, Masayuki, additional, Takahashi, Masanori, additional, Taichi, Namie, additional, Hasuike, Tomoya, additional, Higuchi, Eriko, additional, Kobayashi, Hisako, additional, Osakada, Kaori, additional, Tsuda, Emiko, additional, Shimohama, Shun, additional, Hayashi, Takashi, additional, Hisahara, Shin, additional, Kawamata, Jun, additional, Murahara, Takashi, additional, Saitoh, Masaki, additional, Suzuki, Shuichiro, additional, Yamamoto, Daisuke, additional, Ishiyama, Yoko, additional, Ishiyama, Naoko, additional, Noshiro, Mayuko, additional, Takeyama, Rumi, additional, Uwasa, Kaori, additional, Yasuda, Ikuko, additional, van der Kooi, Anneke, additional, de Visser, Marianne, additional, Gibson, Tamar, additional, Casasnovas, Carlos, additional, Alberti Aguilo, Maria Antonia, additional, Homedes-Pedret, Christian, additional, Julia Palacios, Natalia, additional, Diez Porras, Laura, additional, Velez Santamaria, Valentina, additional, Lazaro, Ana, additional, Diez Tejedor, Exuperio, additional, Gomez Salcedo, Pilar, additional, Fernandez-Fournier, Mireya, additional, Lopez Ruiz, Pedro, additional, Rodriguez de Rivera, Francisco Javier, additional, Sastre, Maria, additional, Gamez, Josep, additional, Sune, Pilar, additional, Salvado, Maria, additional, Gili, Gisela, additional, Mazuela, Gonzalo, additional, Illa, Isabel, additional, Cortes Vicente, Elena, additional, Diaz-Manera, Jordi, additional, Querol Gutierrez, Luis Antonio, additional, Rojas Garcia, Ricardo, additional, Vidal, Nuria, additional, Arribas-Ibar, Elisabet, additional, Piehl, Fredrik, additional, Hietala, Albert, additional, Bjarbo, Lena, additional, Sengun, Ihsan, additional, Meherremova, Arzu, additional, Ozcelik, Pinar, additional, Balkan, Bengu, additional, Tuga, Celal, additional, Ugur, Muzeyyen, additional, Erdem-Ozdamar, Sevim, additional, Bekircan-Kurt, Can Ebru, additional, Acar, Nazire Pinar, additional, Yilmaz, Ezgi, additional, Caliskan, Yagmur, additional, Orsel, Gulsah, additional, Efendi, Husnu, additional, Aydinlik, Seda, additional, Cavus, Hakan, additional, Kutlu, Ayse, additional, Becerikli, Gulsah, additional, Semiz, Cansu, additional, Tun, Ozlem, additional, Terzi, Murat, additional, Dogan, Baki, additional, Onar, Musa Kazim, additional, Sen, Sedat, additional, Kirbas Cavdar, Tugce, additional, Veske, Adife, additional, Norwood, Fiona, additional, Dimitriou, Aikaterini, additional, Gollogly, Jakit, additional, Mahdi-Rogers, Mohamed, additional, Seddigh, Arshira, additional, Sokratous, Giannis, additional, Maier, Gal, additional, Sohail, Faisal, additional, Jacob, Saiju, additional, Sadalage, Girija, additional, Torane, Pravin, additional, Brown, Claire, additional, Shah, Amna, additional, Sathasivam, Sivakumar, additional, Arndt, Heike, additional, Davies, Debbie, additional, Watling, Dave, additional, Amato, Anthony, additional, Cochrane, Thomas, additional, Salajegheh, Mohammed, additional, Roe, Kristen, additional, Amato, Katherine, additional, Toska, Shirli, additional, Wolfe, Gil, additional, Silvestri, Nicholas, additional, Patrick, Kara, additional, Zakalik, Karen, additional, Katz, Jonathan, additional, Miller, Robert, additional, Engel, Marguerite, additional, Forshew, Dallas, additional, Bravver, Elena, additional, Brooks, Benjamin, additional, Plevka, Sarah, additional, Burdette, Maryanne, additional, Cunningham, Scott, additional, Sanjak, Mohammad, additional, Kramer, Megan, additional, Nemeth, Joanne, additional, Schommer, Clara, additional, Tierney, Scott, additional, Juel, Vern, additional, Guptill, Jeffrey, additional, Hobson-Webb, Lisa, additional, Massey, Janice, additional, Beck, Kate, additional, Carnes, Donna, additional, Loor, John, additional, Anderson, Amanda, additional, Pascuzzi, Robert, additional, Bodkin, Cynthia, additional, Kincaid, John, additional, Snook, Riley, additional, Guingrich, Sandra, additional, Micheels, Angela, additional, Chaudhry, Vinay, additional, Corse, Andrea, additional, Mosmiller, Betsy, additional, Kelley, Andrea, additional, Ho, Doreen, additional, Srinivasan, Jayashri, additional, Vytopil, Michal, additional, Jara, Jordan, additional, Ventura, Nicholas, additional, Scala, Stephanie, additional, Carter, Cynthia, additional, Donahue, Craig, additional, Herbert, Carol, additional, Weiner, Elaine, additional, Alam, Sharmeen, additional, McKinnon, Jonathan, additional, Haar, Laura, additional, McKinnon, Naya, additional, Alcon, Karan, additional, McKenna, Kaitlyn, additional, Sattar, Nadia, additional, Daniels, Kevin, additional, Jeffery, Dennis, additional, Freimer, Miriam, additional, Hoyle, Joseph Chad, additional, Kissel, John, additional, Agriesti, Julie, additional, Chelnick, Sharon, additional, Mezache, Louisa, additional, Pineda, Colleen, additional, Muharrem, Filiz, additional, Karam, Chafic, additional, Khoury, Julie, additional, Marburger, Tessa, additional, Kaur, Harpreet, additional, Dimitrova, Diana, additional, Gilchrist, James, additional, Agrawal, Brajesh, additional, Elsayed, Mona, additional, Kohlrus, Stephanie, additional, Andoin, Angela, additional, Darnell, Taylor, additional, Golden, Laura, additional, Lokaitis, Barbara, additional, Seelback, Jenna, additional, Muppidi, Srikanth, additional, Goyal, Neelam, additional, Sakamuri, Sarada, additional, So, Yuen T., additional, Paulose, Shirley, additional, Pol, Sabrina, additional, Welsh, Lesly, additional, Bhavaraju-Sanka, Ratna, additional, Tobon Gonzales, Alejandro, additional, Dishman, Lorraine, additional, Jones, Floyd, additional, Gonzalez, Anna, additional, Padilla, Patricia, additional, Saklad, Amy, additional, Silva, Marcela, additional, Kazamel, Mohamed, additional, Alsharabati, Mohammad, additional, Lu, Liang, additional, Nozaki, Kenkichi, additional, Mumfrey-Thomas, Sandi, additional, Woodall, Amy, additional, Mozaffar, Tahseen, additional, Cash, Tiyonnoh, additional, Goyal, Namita, additional, Roy, Gulmohor, additional, Mathew, Veena, additional, Maqsood, Fatima, additional, Minton, Brian, additional, Jones, H. James, additional, Rosenfeld, Jeffrey, additional, Garcia, Rebekah, additional, Echevarria, Laura, additional, Garcia, Sonia, additional, Pulley, Michael, additional, Aranke, Shachie, additional, Berger, Alan Ross, additional, Shah, Jaimin, additional, Shabbir, Yasmeen, additional, Smith, Lisa, additional, Varghese, Mary, additional, Gutmann, Laurie, additional, Gutmann, Ludwig, additional, Jerath, Nivedita, additional, Nance, Christopher, additional, Swenson, Andrea, additional, Olalde, Heena, additional, Kressin, Nicole, additional, Sieren, Jeri, additional, Barohn, Richard, additional, Dimachkie, Mazen, additional, Glenn, Melanie, additional, McVey, April, additional, Pasnoor, Mamatha, additional, Statland, Jeffery, additional, Wang, Yunxia, additional, Liu, Tina, additional, Emmons, Kelley, additional, Jenci, Nicole, additional, Locheke, Jerry, additional, Fondaw, Alex, additional, Johns, Kathryn, additional, Rico, Gabrielle, additional, Walsh, Maureen, additional, Herbelin, Laura, additional, Hafer-Macko, Charlene, additional, Kwan, Justin, additional, Zilliox, Lindsay, additional, Callison, Karen, additional, Young, Valerie, additional, DiSanzo, Beth, additional, Naunton, Kerry, additional, Benatar, Michael, additional, Bilsker, Martin, additional, Sharma, Khema, additional, Cooley, Anne, additional, Reyes, Eliana, additional, Michon, Sara-Claude, additional, Sheldon, Danielle, additional, Steele, Julie, additional, Howard Jr, James, additional, Chopra, Manisha, additional, Traub, Rebecca, additional, Vu, Tuan, additional, Katzin, Lara, additional, McClain, Terry, additional, Harvey, Brittany, additional, Hart, Adam, additional, Huynh, Kristin, additional, Beydoun, Said, additional, Chilingaryan, Amaiak, additional, Doan, Victor, additional, Droker, Brian, additional, Gong, Hui, additional, Karimi, Sanaz, additional, Lin, Frank, additional, Pokala, Krishna, additional, Shah, Akshay, additional, Tran, Anh, additional, Akhter, Salma, additional, Malekniazi, Ali, additional, Tandan, Rup, additional, Hehir, Michael, additional, Waheed, Waqar, additional, Lucy, Shannon, additional, Weiss, Michael, additional, Distad, Jane, additional, Strom, Susan, additional, Downing, Sharon, additional, Kim, Bryan, additional, Nowak, Richard, additional, Dicapua, Daniel, additional, Keung, Benison, additional, Kumar, Aditya, additional, Patwa, Huned, additional, Robeson, Kimberly, additional, Yang, Irene, additional, Nye, Joan, additional, and Vu, Hong, additional

Published
2019
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49. FM19G11 preserves blood-brain barrier structural and functional integrity by reducing astrocyte toxicity in a human-derived in vitro model of amyotrophic lateral sclerosis

Author

BONANNO, SILVIA and Bonanno, S

Subjects
MED/26 - NEUROLOGIA, Astrociti, FM19G11, SLA, ALS, Astrocyte, BEE, BBB, Farmacoresistenza
Abstract

La Sclerosi Laterale Amiotrofica (SLA) è una patologia neurodegenerativa progressiva in cui le disfunzioni della barriera emato-encefalica (BEE) contribuiscono alla patogenesi e limitano significativamente lo sviluppo di nuove terapie. Gli astrociti di pazienti affetti da forme familiari e sporadiche di SLA rilasciano fattori solubili implicati nel danno della BEE e nell’aumento della P-glicoproteina (P-gp), la principale pompa dell’efflusso di farmaci espressa dalle cellule endoteliali nel cervello, responsabile dei fenomeni di progressiva farmacoresistenza nella SLA. Recentemente è stato dimostrato che FM19G11, un nuovo composto chimico, limita l’attivazione astrocitaria in un modello di lesione del midollo spinale, riducendo la risposta cellulare allo stress. Lo scopo di questa tesi era esaminare gli effetti del trattamento con FM19G11 sugli astrociti reattivi di pazienti SLA a livello della BEE. Abbiamo quindi realizzato un modello in vitro di BEE derivato da cellule staminali umane pluripotenti indotte (hiPSC) e composto da cellule endoteliali controllo (EC) in co-coltura con astrociti ottenuti da iPSC di un soggetto controllo, un paziente SLA familiare con mutazione SOD1-A4V e un soggetto con SLA sporadica. Gli astrociti venivano trattati con FM19G11 per 48h. Parallelamente, le cellule endoteliali erano coltivate su una membrana porosa all’interno di inserti affinchè formassero il monostrato della barriera. Dopo 48h il terreno di coltura degli astrociti veniva sostituito con nuovo terreno e gli inserti con le cellule endoteliali (rappresentanti il “lato del circolo ematico” della BEE) venivano trasferite al di sopra delle colture di astrociti ( il “lato encefalico” della BBB). Le co-colture, che garantivano la contiguità tra i surnatanti evitando il contatto diretto tra le diverse popolazioni cellulari, venivano protratte per 5 giorni. La formazione e l’integrità del monostrato di cellule endoteliali veniva valutato tramite: immunocitochimica per Zonula Occludens-1 (ZO-1), una delle principali proteine costituenti le giunzioni serrate presenti tra le cellule endoteliali nel cervello; misurazione della resistenza elettrica trans-endoteliale (TEER); analisi della permeabilità di fluoresceina sodica (NaF), una molecola solubile in acqua che attraversa la barriera solo tramite diffusione paracellulare, indicatore quindi dell’integrità delle giunzioni tra le cellule endoteliali. Al fine di valutare la presenza del fenomeni di farmacoresistenza, abbiamo misurato l’attività di trasporto di P-gp tramite l’efflusso di Rodamina (Rh123), un substrato specifico di P-gp. Inoltre, l’espressione di P-gp da parte delle cellule endoteliali è stata analizzata tramite immunocitochimica e immunofissazione. Per valutare le vie molecolari sottese alla modulazione delle cellule endoteliali abbiamo calcolato il rapporto tra espressione nucleare e citoplasmatica del fattore NF-kB tramite analisi di immunocitochimica. Inoltre, abbiamo analizzato, tramite specifiche metodiche fluorometriche, l’effetto di FM19G11 sui possibili mediatori dell’attivazione di NF-kB rilasciati dagli astrociti reattivi da pazienti SLA, le specie reattive dell’ossigeno (ROS) e il glutammato. Rispetto alle co-colture composte da astrociti non trattati, le cellule endoteliali coltivate con gli astrociti SOD1-A4V a sporadici trattati con FM19G11 hanno mostrato: ì) una maggiore resistenza, congrua con una minore permeabilità, del monostrato; ii) una ridotta espressione e funzione di efflusso di P-gp; iii) una ridotta attivazione di NF-kB, in linea con la minore produzione di ROS e glutammato da parte degli astrociti SOD1-A4V e sporadici trattati con FM19G11. FM19G11 mantiene l’integrità della BEE e riduce la sovra espressione di P-gp ,diminuendo la reattività astrocitaria, pertanto potrebbe essere considerato per futuri approcci terapeutici combinatoriali per la terapia della SLA. Amyotrophic lateral sclerosis (ALS) is a progressive neurodegenerative disease in which blood brain barrier (BBB) dysfunction contributes to the pathogenesis and significantly limits the development of successful therapies. Astrocytes from familial and sporadic ALS patients release soluble factors implicated in BBB injury and upregulation of P-glycoprotein (P-gp), the main multidrug efflux transporter in brain endothelial cells, responsible for progressive pharmacoresistance in ALS. Recently, FM19G11, a new chemical compound, was shown to counteract astrogliosis in a spinal cord injury model, decreasing cell stress response in the microenvironment. Specific objectives of this thesis were: i) to evaluate the potential effect of FM19G11 treatment on astrocyte-driven BBB dysruption and pharmacoresistance; ii) to identify ALS astrocyte molecular features targeted by FM19G11 compound. To this purpose, we set up a human induced pluripotent stem cells (hiPSCs)-derived BBB in vitro system composed of control iPS-endothelial cells (ECs), co-cultured with hiPSCs-derived astrocytes from control, familial SOD1-A4V and Sporadic ALS patients. Control and ALS-astrocytes were conditioned with FM19G11 500nM for 48h. In parallel, ECs were seeded on transwell porous membrane inserts, allowing to form the endothelial monolayer. After 48h, astrocyte culture medium was replaced with fresh medium, and ECs-inserts (representing the “blood side” of the BBB) were placed atop of the pre-treated astrocyte layer (representing the “brain side”). Co-cultures in the transwell culture system, which guaranteed supernatant interaction, avoiding cell contact, were maintained for 5 days. Formation and integrity of human iPSC endothelial monolayer were examined by: immunostaining for Zonula Occludens-1 (ZO-1), a major protein constituent of brain endothelial tight junctions (TJs); trans-endothelial electric resistance (TEER) assessment; sodium fluorescein (NaF) permeability assay, a water-soluble molecule that cross the BBB only through paracellular diffusion, thus being an indicator of endothelial cell-to-cell cohesion. In order to investigate pharmacoresistance occurrence, we determined P-gp transport activity by calculating Rhodamine 123 (Rh123), a P-gp specific substrate, efflux ratio. P-gP expression was assessed on endothelial monolayers by immunocytochemistry and western-blot analysis. To examine the molecular signaling underlying endothelial cells modulation, nuclear factor kappa-light-chain-enhancer of activated B cells (NF-kB) nuclear to cytoplasmic ratio was investigated by immunocytochemical expression analysis. Further, we examined FM19G11 effect on potential NF-kB upstream mediators released by reactive ALS astrocytes. Hence, astrocyte-derived reactive oxygen species (ROS) and glutamate levels in astrocyte conditioned medium were also measured by highly sensitive fluorometric approaches. Compared to co-cultures composed by untreated ALS-astrocytes, endothelial monolayers co-cultured with FM19G11-conditioned SOD1-A4V and sporadic astrocytes showed: i) an improvement in endothelial monolayer resistance, compatible with a decreased passive permeability across the BBB in vitro model; ii) a consistently lower P-gp expression and efflux activity; ììì) a reduced NF-kB activation, in line with the detected decreased ROS and glutamate production by FM19G11-treated SOD1-A4V and sporadic astrocytes. FM19G11 preserves BBB integrity, and restrains P-gP overexpression, by reducing cell stress factors released from familial SOD1-A4V and sporadic ALS astrocyte in a human-derived in vitro cell model. The outcome of our investigation suggests that FM19G11 is able to limit BBB dysfunction and could impact brain accessibility of therapeutics thus, it might be considered for future combinatorial therapeutic strategies for ALS.

Published
2019

50. Long-term safety and efficacy of eculizumab in generalized myasthenia gravis

Author

Muppidi, Srikanth, Utsugisawa, Kimiaki, Benatar, Michael, Murai, Hiroyuki, Barohn, Richard J., Illa, Isabel, Jacob, Saiju, Vissing, John, Burns, Ted M., Kissel, John T., Nowak, Richard J., Andersen, Henning, Casasnovas, Carlos, de Bleecker, Jan L., Vu, Tuan H., Mantegazza, Renato, O'Brien, Fanny L., Wang, Jing Jing, Fujita, Kenji P., Howard, James F., Mazia, Claudio Gabriel, Wilken, Miguel, Barroso, Fabio, Saba, Juliet, Rugiero, Marcelo, Bettini, Mariela, Chaves, Marcelo, Vidal, Gonzalo, Garcia, Alejandra Dalila, van den Abeele, Guy, de Koning, Kathy, de Mey, Katrien, Mercelis, Rudy, Mahieu, D. lphine, Wagemaekers, Linda, van Damme, Philip, Depreitere, Annelies, Schotte, Caroline, Smetcoren, Charlotte, Stevens, Olivier, van Daele, Sien, Vandenbussche, Nicolas, Vanhee, Annelies, Verjans, Sarah, Vynckier, Jan, D'Hondt, Ann, Tilkin, Petra, Alves de Siqueira Carvalho, Alzira, Dias Brockhausen, Igor, Feder, David, Ambrosio, Daniel, César, Pamela, Melo, Ana Paula, Martins Ribeiro, Renata, Rocha, Rosana, Bezerra Rosa, Bruno, Veiga, Thabata, da Silva, Luiz Augusto, Santos Engel, Murilo, Gonçalves Geraldo, Jordana, Ananias Morita, Maria da Penha, Nogueira Coelho, Erica, Paiva, Gabriel, Pozo, Marina, Prando, Natalia, Martineli Torres, Debora Dada, Butinhao, Cristiani Fernanda, Duran, Gustavo, Gomes da Silva, Tamires Cristina, Otavio Maia Gonçalves, Luiz, Pazetto, Lucas Eduardo, Fialho, Tomás Augusto Suriane, Renata Cubas Volpe, Luciana, Souza Duca, Luciana, Gheller Friedrich, Maurício André, Guerreiro, Alexandre, Mohr, Henrique, Pereira Martins, Maurer, da Cruz Pacheco, Daiane, Ferreira, Luciana, Macagnan, Ana Paula, Pinto, Graziela, de Cassia Santos, Aline, Souza Bulle Oliveira, Acary, Amaral Andrade, Ana Carolina, Annes, Marcelo, Duarte Silva, Liene, Cavalcante Lino, Valeria, Pinto, Wladimir, Assis, Natália, Carrara, Fernanda, Miranda, Carolina, Souza, Iandra, Fernandes, Patricia, Siddiqi, Zaeem, Phan, Cecile, Narayan, Jeffrey, Blackmore, Derrick, Mallon, Ashley, Roderus, Rikki, Watt, Elizabeth, Vohanka, Stanislav, Bednarik, Josef, Chmelikova, Magda, Cierny, Marek, Toncrova, Stanislava, Junkerova, Jana, Kurkova, Barbora, Reguliova, Katarina, Zapletalova, Olga, Pitha, Jiri, Novakova, Iveta, Tyblova, Michaela, Jurajdova, Ivana, Wolfova, Marcela, Harbo, Thomas, Vinge, Lotte, Krogh, Susanne, Mogensen, Anita, Højgaard, Joan, Witting, Nanna, Ostergaard Autzen, Anne, Pedersen, Jane, Eralinna, Juha-Pekka, Laaksonen, Mikko, Oksaranta, Olli, Harrison, Tuula, Eriksson, Jaana, Rozsa, Csilla, Horvath, Melinda, Lovas, Gabor, Matolcsi, Judit, Szabo, Gyorgyi, Jakab, Gedeonne, Szabadosne, Brigitta, Vecsei, Laszlo, Dezsi, Livia, Varga, Edina, Konyane, Monika, Antonini, Giovanni, di Pasquale, Antonella, Garibaldi, Matteo, Morino, Stefania, Troili, Fernanda, Fionda, Laura, Filla, Allessandro, Costabile, Teresa, Marano, Enrico, Saccà, Francesco, Fasanaro, Angiola, Marsili, Angela, Puorro, Giorgia, Antozzi, Carlo, Bonanno, Silvia, Camera, Giorgia, Locatelli, Alberta, Maggi, Lorenzo, Pasanisi, Maria, Campanella, Angela, Evoli, Amelia, Alboini, Paolo Emilio, D'Amato, Valentina, Iorio, Raffaele, Inghilleri, Maurizio, Frasca, Vittorio, Giacomelli, Elena, Gori, Maria, Lopergolo, Diego, Onesti, Emanuela, Gabriele, Maria, Uzawa, Akiyuki, Kanai, Tetsuya, Kawaguchi, Naoki, Mori, Masahiro, Kaneko, Yoko, Kanzaki, Akiko, Kobayashi, Eri, Masaki, Katsuhisa, Matsuse, Dai, Matsushita, Takuya, Uehara, Taira, Shimpo, Misa, Jingu, Maki, Kikutake, Keiko, Nakamura, Yumiko, Sano, Yoshiko, Nagane, Yuriko, Kamegamori, Ikuko, Tsuda, Tomoko, Fujii, Yuko, Futono, Kazumi, Ozawa, Yukiko, Mizugami, Aya, Saito, Yuka, Suzuki, Hidekazu, Morikawa, Miyuki, Samukawa, Makoto, Kamakura, Sachiko, Miyawaki, Eriko, Shiraishi, Hirokazu, Mitazaki, Teiichiro, Motomura, Masakatsu, Mukaino, Akihiro, Yoshimura, Shunsuke, Asada, Shizuka, Yoshida, Seiko, Amamoto, Shoko, Kobashikawa, Tomomi, Koga, Megumi, Maeda, Yasuko, Takada, Kazumi, Takada, Mihoko, Tsurumaru, Masako, Yamashita, Yumi, Suzuki, Yasushi, Akiyama, Tetsuya, Narikawa, Koichi, Tano, Ohito, Tsukita, Kenichi, Kurihara, Rikako, Meguro, Fumie, Fukuda, Yusuke, Sato, Miwako, Okumura, Meinoshin, Funaka, Soichiro, Kawamura, Tomohiro, Makamori, Masayuki, Takahashi, Masanori, Taichi, Namie, Hasuike, Tomoya, Higuchi, Eriko, Kobayashi, Hisako, Osakada, Kaori, Imai, Tomihiro, Tsuda, Emiko, Shimohama, Shun, Hayashi, Takashi, Hisahara, Shin, Kawamata, Jun, Murahara, Takashi, Saitoh, Masaki, Suzuki, Shuichiro, Yamamoto, Daisuke, Ishiyama, Yoko, Ishiyama, Naoko, Noshiro, Mayuko, Takeyama, Rumi, Uwasa, Kaori, Yasuda, Ikuko, van der Kooi, Anneke, de Visser, Marianne, Gibson, Tamar, Kim, Byung-Jo, Lee, Chang Nyoung, Koo, Yong Seo, Seok, Hung Youl, Kang, Hoo Nam, Ra, HyeJin, Kim, Byoung Joon, Cho, Eun Bin, Choi, MiSong, Lee, HyeLim, Min, Ju-Hong, Seok, Jinmyoung, Lee, JiEun, Koh, Da Yoon, Kwon, JuYoung, Park, SangAe, Choi, Eun Hwa, Hong, Yoon-Ho, Ahn, So-Hyun, Koo, Dae Lim, Lim, Jae-Sung, Shin, Chae Won, Hwang, Ji Ye, Kim, Miri, Kim, Seung Min, Jeong, Ha-Neul, Jung, JinWoo, Kim, Yool-hee, Lee, Hyung Seok, Shin, Ha Young, Hwang, Eun Bi, Shin, Miju, Alberti Aguilo, Maria Antonia, Homedes-Pedret, Christian, Julia Palacios, Natalia, Diez Porras, Laura, Velez Santamaria, Valentina, Lazaro, Ana, Diez Tejedor, Exuperio, Gomez Salcedo, Pilar, Fernandez-Fournier, Mireya, Lopez Ruiz, Pedro, Rodriguez de Rivera, Francisco Javier, Sastre, Maria, Gamez, Josep, Sune, Pilar, Salvado, Maria, Gili, Gisela, Mazuela, Gonzalo, Cortes Vicente, Elena, Diaz-Manera, Jordi, Querol Gutierrez, Luis Antonio, Rojas Garcia, Ricardo, Vidal, Nuria, Arribas-Ibar, Elisabet, Piehl, Fredrik, Hietala, Albert, Bjarbo, Lena, Sengun, Ihsan, Meherremova, Arzu, Ozcelik, Pinar, Balkan, Bengu, Tuga, Celal, Ugur, Muzeyyen, Erdem-Ozdamar, Sevim, Bekircan-Kurt, Can Ebru, Acar, Nazire Pinar, Yilmaz, Ezgi, Caliskan, Yagmur, Orsel, Gulsah, Efendi, Husnu, Aydinlik, Seda, Cavus, Hakan, Kutlu, Ayse, Becerikli, Gulsar, Semiz, Cansu, Tun, Ozlem, Terzi, Murat, Dogan, Baki, Onar, Musa Kazim, Sen, Sedat, Kirbas Cavdar, Tugce, Veske, Adife, Norwood, Fiona, Dimitriou, Aikaterini, Gollogly, Jakit, Mahdi-Rogers, Mohamed, Seddigh, Arshira, Sokratous, Giannis, Maier, Gal, Sohail, Faisal, Sadalage, Girija, Torane, Pravin, Brown, Claire, Shah, Amna, Sathasivam, Sivakumar, Arndt, Heike, Davies, Debbie, Watling, Dave, Amato, Anthony, Cochrane, Thomas, Salajegheh, Mohammed, Roe, Kristen, Amato, Katherine, Toska, Shirli, Wolfe, Gil, Silvestri, Nicholas, Patrick, Kara, Zakalik, Karen, Katz, Jonathan, Miller, Robert, Engel, Marguerite, Forshew, Dallas, Bravver, Elena, Brooks, Benjamin, Plevka, Sarah, Burdette, Maryanne, Cunningham, Scott, Sanjak, Mohammad, Kramer, Megan, Nemeth, Joanne, Schommer, Clara, Tierney, Scott, Juel, Vern, Guptill, Jeffrey, Hobson-Webb, Lisa, Massey, Janice, Beck, Kate, Carnes, Donna, Loor, John, Anderson, Amanda, Pascuzzi, Robert, Bodkin, Cynthia, Kincaid, John, Snook, Riley, Guingrich, Sandra, Micheels, Angela, Chaudhry, Vinay, Corse, Andrea, Mosmiller, Betsy, Kelley, Andrea, Ho, Doreen, Srinivasan, Jayashri, Vytopil, Michal, Jara, Jordan, Ventura, Nicholas, Scala, Stephanie, Carter, Cynthia, Donahue, Craig, Herbert, Carol, Weiner, Elaine, Alam, Sharmeen, McKinnon, Jonathan, Haar, Laura, McKinnon, Naya, Alcon, Karan, McKenna, Kaitlyn, Sattar, Nadia, Daniels, Kevin, Jeffery, Dennis, Freimer, Miriam, Hoyle, Joseph Chad, Agriesti, Julie, Chelnick, Sharon, Mezache, Louisa, Pineda, Colleen, Muharrem, Filiz, Karam, Chafic, Khoury, Julie, Marburger, Tessa, Kaur, Harpreet, Dimitrova, Diana, Gilchrist, James, Agrawal, Brajesh, Elsayed, Mona, Kohlrus, Stephanie, Andoin, Angela, Darnell, Taylor, Golden, Laura, Lokaitis, Barbara, Seelback, Jenna, Goyal, Neelam, Sakamuri, Sarada, So, Yuen T., Paulose, Shirley, Pol, Sabrina, Welsh, Lesly, Bhavaraju-Sanka, Ratna, Tobon Gonzales, Alejandro, Dishman, Lorraine, Jones, Floyd, Gonzalez, Anna, Padilla, Patricia, Saklad, Amy, Silva, Marcela, Nations, Sharon, Trivedi, Jaya, Hopkins, Steve, Kazamel, Mohamed, Alsharabati, Mohammad, Lu, Liang, Nozaki, Kenkichi, Mumfrey-Thomas, Sandi, Woodall, Amy, Mozaffar, Tahseen, Cash, Tiyonnoh, Goyal, Namita, Roy, Gulmohor, Mathew, Veena, Maqsood, Fatima, Minton, Brian, Jones, H. James, Rosenfeld, Jeffrey, Garcia, Rebekah, Echevarria, Laura, Garcia, Sonia, Pulley, Michael, Aranke, Shachie, Berger, Alan Ross, Shah, Jaimin, Shabbir, Yasmeen, Smith, Lisa, Varghese, Mary, Gutmann, Laurie, Gutmann, Ludwig, Jerath, Nivedita, Nance, Christopher, Swenson, Andrea, Olalde, Heena, Kressin, Nicole, Sieren, Jeri, Dimachkie, Mazen, Glenn, Melanie, McVey, April, Pasnoor, Mamatha, Statland, Jeffery, Wang, Yunxia, Liu, Tina, Emmons, Kelley, Jenci, Nicole, Locheke, Jerry, Fondaw, Alex, Johns, Kathryn, Rico, Gabrielle, Walsh, Maureen, Herbelin, Laura, Hafer-Macko, Charlene, Kwan, Justin, Zilliox, Lindsay, Callison, Karen, Young, Valerie, DiSanzo, Beth, Naunton, Kerry, Bilsker, Martin, Sharma, Khema, Cooley, Anne, Reyes, Eliana, Michon, Sara-Claude, Sheldon, Danielle, Steele, Julie, Chopra, Manisha, Traub, Rebecca, Katzin, Lara, McClain, Terry, Harvey, Brittany, Hart, Adam, Huynh, Kristin, Beydoun, Said, Chilingaryan, Amaiak, Doan, Victor, Droker, Brian, Gong, Hui, Karimi, Sanaz, Lin, Frank, Pokala, Krishna, Shah, Akshay, Tran, Anh, Akhter, Salma, Malekniazi, Ali, Tandan, Rup, Hehir, Michael, Waheed, Waqar, Lucy, Shannon, Weiss, Michael, Distad, Jane, Strom, Susan, Downing, Sharon, Kim, Bryan, Bertorini, Tulio, Arnold, Thomas, Hendersen, Kendrick, Pillai, Rekha, Liu, Ye, Wheeler, Lauren, Hewlett, Jasmine, Vanderhook, Mollie, Dicapua, Daniel, Keung, Benison, Kumar, Aditya, Patwa, Huned, Robeson, Kimberly, Yang, Irene, Nye, Joan, Vu, Hong, Muppidi, S., Utsugisawa, K., Benatar, M., Murai, H., Barohn, R. J., Illa, I., Jacob, S., Vissing, J., Burns, T. M., Kissel, J. T., Nowak, R. J., Andersen, H., Casasnovas, C., de Bleecker, J. L., Vu, T. H., Mantegazza, R., O'Brien, F. L., Wang, J. J., Fujita, K. P., Howard, J. F., Mazia, C. G., Wilken, M., Barroso, F., Saba, J., Rugiero, M., Bettini, M., Chaves, M., Vidal, G., Garcia, A. D., Van den Abeele, G., de Koning, K., De Mey, K., Mercelis, R., Mahieu, D., Wagemaekers, L., Van Damme, P., Depreitere, A., Schotte, C., Smetcoren, C., Stevens, O., Van Daele, S., Vandenbussche, N., Vanhee, A., Verjans, S., Vynckier, J., D'Hondt, A., Tilkin, P., Alves de Siqueira Carvalho, A., Dias Brockhausen, I., Feder, D., Ambrosio, D., Cesar, P., Melo, A. P., Martins Ribeiro, R., Rocha, R., Bezerra Rosa, B., Veiga, T., da Silva, L. A., Santos Engel, M., Goncalves Geraldo, J., Ananias Morita, M. D. P., Nogueira Coelho, E., Paiva, G., Pozo, M., Prando, N., Martineli Torres, D. D., Butinhao, C. F., Duran, G., Gomes da Silva, T. C., Otavio Maia Goncalves, L., Pazetto, L. E., Fialho, T. A. S., Renata Cubas Volpe, L., Souza Duca, L., Gheller Friedrich, M. A., Guerreiro, A., Mohr, H., Pereira Martins, M., da Cruz Pacheco, D., Ferreira, L., Macagnan, A. P., Pinto, G., de Cassia Santos, A., Souza Bulle Oliveira, A., Amaral Andrade, A. 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Subjects
0301 basic medicine, Male, Pediatrics, Exacerbation, Physiology, Heart Diseases/chemically induced, Myasthenia Gravis/drug therapy, 030105 genetics & heredity, THERAPY, DOUBLE-BLIND, 0302 clinical medicine, Quality of life, Monoclonal, Activities of Daily Living, Medicine and Health Sciences, Functional ability, Longitudinal Studies, Humanized, Angioedema/chemically induced, MG-ADL, QMG, Eculizumab, Middle Aged, myasthenia gravi, 3. Good health, Treatment Outcome, Antibodies, Monoclonal, Humanized/therapeutic use, Meningococcal Infections/epidemiology, Disease Progression, Female, eculizumab, Life Sciences & Biomedicine, COMPLEMENT INHIBITOR ECULIZUMAB, medicine.drug, Adult, medicine.medical_specialty, MG-QOL15, Heart Diseases, Clinical Neurology, Meningococcal Vaccines, Antibodies, Monoclonal, Humanized, Placebo, Antibodies, ACETYLCHOLINE-RECEPTOR, Complement Inactivating Agents/therapeutic use, 03 medical and health sciences, Cellular and Molecular Neuroscience, Refractory, Physiology (medical), Injection Site Reaction/epidemiology, Myasthenia Gravis, medicine, Aspergillosis, Aspergillosis/epidemiology, Humans, Muscle Strength, Angioedema, myasthenia gravis, Science & Technology, business.industry, MGC, Neurosciences, Meningococcal Vaccines/therapeutic use, medicine.disease, Interim analysis, Complement Inactivating Agents, Injection Site Reaction, Meningococcal Infections, Quality of Life, Myasthenia gravis, ANTIBODY, Neurosciences & Neurology, Neurology (clinical), business, 030217 neurology & neurosurgery
Abstract

INTRODUCTION: Eculizumab is effective and well tolerated in patients with antiacetylcholine receptor antibody-positive refractory generalized myasthenia gravis (gMG; REGAIN; NCT01997229). We report an interim analysis of an open-label extension of REGAIN, evaluating eculizumab's long-term safety and efficacy. METHODS: Eculizumab (1,200 mg every 2 weeks for 22.7 months [median]) was administered to 117 patients. RESULTS: The safety profile of eculizumab was consistent with REGAIN; no cases of meningococcal infection were reported during the interim analysis period. Myasthenia gravis exacerbation rate was reduced by 75% from the year before REGAIN (P

Published
2019

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