Cognitive decline in Huntington's disease expansion gene carriers

Anna Rita Bentivoglio, Verena Baake, Robert H.A.M. Reijntjes, Eve M. Dumas, Jennifer C. Thompson, Raymund A.C. Roos, Jirí Klempír, Ruth Fullam, Raphael M. Bonelli, Karen Hecht, Brigitte Herranhof, Anna Holl, Hans-Peter Kapfhammer, Michael Koppitz, Sabine Lilek, Markus Magnet, Nicole Müller, Daniela Otti, Annamaria Painold, Karin ReisingerMonika Scheibl, Helmut Schöggl, Jasmin Ullah, Eva-Maria Braunwarth, Florian Brugger, Lisa Buratti, Eva-Maria Hametner, Caroline Hepperger, Christiane Holas, Anna Hotter, Anna Hussl, Barbara Larcher, Philipp Mahlknecht, Christoph Müller, Bernadette Pinter, Werner Poewe, Eva-Magdalena Reiter, Klaus Seppi, Fabienne Sprenger, Gregor Wenning, Gunther Ladurner, Stefan Lilek, Daniela Sinadinosa, Wolfgang Staffen, Anna Maria Walleczek, Michel Dupuis, Cécile Minet, Pascale Ribaï, Dominique Van Paemel, Christine Verellen-Dumoulin, Veronika Majerová, Jan Roth, Paivi Hartikainen, Mari Ollokainen, Maire Santala, Pia Eklund, Heli Hiivola, Hannele Hyppönen, Kirsti Martikainen, Katri Tuuha, Philippe Allain, Dominique Bonneau, Marie Bost, Bénédicte Gohier, Marie-Anne Guérid, Audrey Olivier, Julie Prouzet, Adriana Prundean, Clarisse Scherer-Gagou, Christophe Verny, Blandine Babiloni, Sabrina Debruxelles, Charlotte Duché, Cyril Goizet, Laetitia Jameau, Danielle Lafoucrière, Umberto Spampinato, Julien Couttier, Bérengère Debilly, Christine Delaigue, Franck Durif, Perrine Legendre, Sylvie Loiseau, Miguel Ulla, Tiphaine Vidal, Anne-Catherine Bachoud-Lévi, Farideh Badei, Marie-Françoise Boissé, Lotfi Boudali, Laurent Cleret De Langavant, Laurie Lemoine, Graca Morgado, Katia Youssov, Agnès Annic, Recka Barthélémy, Christelle De Bruycker, Maryline Cabaret, Anne-Sophie Carette, Nicolas Carrière, Eric Decorte, Luc Defebvre, Marie Delliaux, Arnaud Delval, Alizé Depelchin, Alain Destee, Nelly Dewulf-Pasz, Thibaut Dondaine, Florence Dugauquier, Kathy Dujardin, Lucie Hopes, Pierre Krystkowiak, Marie-Helene Lemaire, Sylvie Manouvrier, Eugénie Mutez, Mireille Peter, Lucie Plomhause, Bernard Sablonnière, Clémence Simonin, Céline Tard, Stéphanie Thibault-Tanchou, Isabelle Vuillaume, Marcellin Bellonet, Alexandra Benoit, Hassan Berrisoul, Stéphanie Blin, Françoise Courtin, Cécile Duru, Véronique Fasquel, Mélanie Flament, Olivier Godefroy, Béatrice Mantaux, Alicia Playe, Martine Roussel, Mélissa Tir, Béatrice Schüler, Sandrine Wannepain, Jean-Philippe Azulay, Christelle Chabot, Marie Delfini, Alexandre Eusebio, Frédérique Fluchere, Christine Garreau, Hélène Grosjean, Aicha Guenam, Laura Mundler, Marielle Nowak, Rolland Raseta, Sandra Benaich, Alexis Brice, Sarah Boster, Perrine Charles, Alexandra Durr, Claire Ewenczyk, Hélène Francisque, Céline Jauffret, Damian Justo, Abdulrahman Kassar, Stephan Klebe, Fabien Lesne, Paolo Milani, Marie-Lorraine Monin, Emmanuel Roze, Alina Tataru, Maya Tchikviladzé, Sandrine Bioux, Evangeline Bliaux, Carole Girard, Lucie Guyant-Maréchal, Didier Hannequin, Véronique Hannier, Séverine Jourdain, David Maltête, Dorothée Pouliquen, Mathieu Anheim, Nadia Barun, Ouhaid Lagha-Boukbiza, Nadine Longato, Christophe Marcel, Clélie Phillipps, Gabrielle Rudolf, Gisèle Steinmetz, Christine Tranchant, Caroline Wagner, Marie-Agathe Zimmermann, Leily Blondeau, Fabienne Calvas, Samia Cheriet, Helène Delabaere, Jean-François Demonet, Jérémie Pariente, Michèle Pierre, Sandrine Rolland, Christoph Michael Kosinski, Eva Milkereit, Daniela Probst, Kathrin Reetz, Christian Sass, Johannes Schiefer, Christiane Schlangen, Cornelius J. Werner, Markus Beuth, Harald Gelderblom, Josef Priller, Harald Prüß, Eike Spruth, Silvia Thiel, Jürgen Andrich, Gisa Ellrichmann, Lennard Herrmann, Rainer Hoffmann, Barbara Kaminski, Peter Kraus, Carsten Saft, Christiane Stamm, Herwig Lange, Robert Maiwald, Cecile Bosredon, Ulrike Hunger, Matthias Löhle, Antonia Maass, Christiana Ossig, Simone Schmidt, Alexander Storch, Annett Wolz, Martin Wolz, Zacharias Kohl, Christina Kozay, Jürgen Winkler, Ulrike Bergmann, Regina Böringer, Philipp Capetian, Gerit Kammel, Johann Lambeck, Miriam Mächtel, Simone Meier, Michel Rijntjes, Birgit Zucker, Kai Boelmans, Christos Ganos, Ines Goerendt, Walburgis Heinicke, Ute Hidding, Jan Lewerenz, Alexander Münchau, Michael Orth, Jenny Schmalfeld, Lars Stubbe, Simone Zittel, Gabriele Diercks, Dirk Dressler, Flverly Francis, Sabine Gayde-Stephan, Heike Gorzolla, Bianca Kramer, Rebecca Minschke, Christoph Schrader, Pawel Tacik, Michael Ribbat, Bernhard Longinus, Carsten Möller, Katrin Bürk, Antje Lüsebrink, Mark Mühlau, Alexander Peinemann, Michael Städtler, Adolf Weindl, Juliane Winkelmann, Cornelia Ziegler, Natalie Bechtel, Heike Beckmann, Stefan Bohlen, Nicole Göpfert, Eva Hölzner, Ralf Reilmann, Stefanie Rohm, Silke Rumpf, Sigrun Schepers, Nathalia Weber, Michael Bachmeier, Matthias Dose, Nina Hofstetter, Ralf Marquard, Alzbeta Mühlbäck, Katrin Barth, Andrea Buck, Julia Connemann, Daniel Ecker, Carolin Geitner, Christine Held, Andrea Kesse, Bernhard Landwehrmeyer, Franziska Lezius, Solveig Nepper, Anke Niess, Ariane Schneider, Daniela Schwenk, Sigurd Süssmuth, Sonja Trautmann, Melanie Vogel, Patrick Weydt, Claudia Cormio, Olimpia Difruscolo, Giovanni Franco, Vittorio Sciruicchio, Claudia Serpino, Marina De Tommaso, Giovanna Calandra-Buonaura, Sabina Capellari, Pietro Cortelli, Roberto Gallassi, Roberto Poda, Cesa Scaglione, Elisabetta Bertini, Caterina Bartoli, Fernanda Fortunato, Elena Ghelli, Andrea Ginestroni, Claudia Mechi, Marco Paganini, Silvia Piacentini, Silvia Pradella, Anna Maria Romoli, Sandro Sorbi, Giovanni Abbruzzese, Monica Bandettini Di Poggio, Giovanna Ferrandes, Paola Mandich, Roberta Marchese, Emilio Di Maria, Simona Castagliuolo, Anna Castaldo, Stefano Di Donato, Daniela Di Bella, Cinzia Gellera, Silvia Genitrini, Caterina Mariotti, Daniela Monza, Lorenzo Nanetti, Marta Panzeri, Dominga Paridi, Paola Soliveri, Francesca Spagnolo, Franco Taroni, Chiara Tomasello, Giuseppe De Michele, Luigi Di Maio, Carlo Rinaldi, Marco Massarelli, Silvio Peluso, Alessandro Roca, Cinzia Valeria Russo, Elena Salvatore, Pierpaolo Sorrentino, Tecla Tucci, Milena Cannella, Valentina Codella, Francesca De Gregorio, Annunziata De Nicola, Francesca Elifani, Chiara Esposito, Tiziana Martino, Irene Mazzante, Martina Petrollini, Maria Simonelli, Maurizio Vezza, Ferdinando Squitieri, Barbara D'Alessio, Francesca Lovo, Giulia Coarelli, Michela Ferraldeschi, Giovanni Ristori, Monique S.E. Van Hout, Jeroen P.P. Van Vugt, A. Marit De Weert, Marloes Verhoeven, Meike Dekker, Nico Leenders, Joost Van Oostrom, Jesper Klooster, Berry Kremer, Simon J.A. Van Den Bogaard, Reineke Bos, Ellen P. T Hart, Anne Kampstra, Anne Schoonderbeek, Annelien Duits, Mayke Oosterloo, Mirella Waber, Ellen Økland Blinkenberg, Erik Hauge, Hilde Tyvoll, Olaf Aaserud, Nils Olaf Aanonsen, Kathrine Bjørgo, Nancy Borgeød, Elisabeth Dramstad, Madeleine Fannemel, Jan C. Frich, Per F. 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Risultato della ricerca: Contributo in rivistaArticolo in rivista

17 Citazioni (Scopus)

Abstract

Background: In Huntington's Disease (HD) cognitive decline can occur before unequivocal motor signs become apparent. As cognitive decline often starts early in the course of the disease and has a progressive nature over time, cognition can be regarded as a key target for symptomatic treatment. The specific progressive profile of cognitive decline over time is unknown.Objective: The aim of this study is to quantify the progression of cognitive decline across all HD stages, from pre-motormanifest to advanced HD, and to investigate if CAG length mediates cognitive decline.Methods: In the European REGISTRY study 2669 HD expansion gene carriers underwent annual cognitive assessment. General linear mixed models were used to model the cognitive decline for each cognitive task across all disease stages. Additionally, a model was developed to evaluate the cognitive decline based on CAG length and age rather than disease stage.Results: There was significant cognitive decline on all administered tasks throughout pre-motormanifest (close to estimated disease onset) participants and the subsequent motormanifest participants from stage 1 to stage 4. Performance on the Stroop Word and Stroop Color tests additionally declined significantly across the two pre-motormanifest groups: far and close to estimated disease onset.The evaluation of cognition performance in relation to CAG length and age revealed a more rapid cognitive decline in participants with longer CAG length than participants with shorter CAG length over time.Conclusion: Cognitive performance already shows decline in pre-motormanifest HD gene expansion carriers and gradually worsens to late stage HD. HD gene expansion carriers with certain CAG length have their own cognitive profile, i.e., longer CAG length is associated with more rapid decline. (C) 2017 Elsevier Ltd. All rights reserved.
Lingua originaleEnglish
pagine (da-a)51-62
Numero di pagine12
RivistaCortex
Volume95
Stato di pubblicazionePubblicato - 2017

Keywords

  • Cognition
  • Huntington's disease
  • Longitudinal
  • REGISTRY

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