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Neurocognitive outcomes in neurofibromatosis clinical trials

2016; Lippincott Williams & Wilkins; Volume: 87; Issue: 7_Supplement_1 Linguagem: Inglês

10.1212/wnl.0000000000002928

1526-632X

Karin S. Walsh, Jennifer Janusz, Pamela L. Wolters, Staci Martin, Bonnie Klein-Tasman, Mary Anne Toledo‐Tamula, Heather L. Thompson, Jonathan M. Payne, Kristina K. Hardy, Peter de Blank, Claire Semerjian, Laura Schaffner Gray, Sondra E. Solomon, Nicole J. Ullrich, Shivani Ahlawat, Srivandana Akshintala, Jeffrey C. Allen, Simone Ardern‐Holmes, Robert A. Avery, Amedeo A. Azizi, Dusica Babovic‐Vuksanovic, Annette Bakker, Andrea Baldwin, Fred G. Barker, Amanda Bergner, Chetan Bettegowda, Sucharita Bhaumik, Larissa T. Bilaniuk, Kim Bischoff, Jaishri Blakely, Diana Bradford, Miriam A. Bredella, Wenli Cai, John Carino, Avneesh Chabra, Patricia Ciavarelli, Wade Clapp, Steve Connor, Albert Cornelius, Tambra Dahlheimer, Stephanie H. Davis, Peter de Blank, Vidya Dhote, Joni K. Doherty, Eva Dombi, William N. Dudley, Rachel Ershler, D. Gareth Evans, Laura M. Fayad, Cristina Fernández‐Valle, Rosalie E. Ferner, Michael J. Fisher, Barbara Franklin, Giulia Fulci, Tracy Galloway, Kathy Gardner, Richard Gedrich, Marco Giovannini, Amy Goldstein, Anne Goodwin, Stéphane Goutagny, David H. Gutmann, Theresa Hadlock, Chris Halpin, C. Oliver Hanemann, Kristina K. Hardy, Gordon J. Harris, Desiree Headley, Gena Heidary, Jonathan Heller, Cynthia M. Hingtgen, Trent R. Hummel, Susan Huson, Michael A. Jacobs, Jennifer Janusz, Diego Jaramillo, Justin T. Jordan, Allen Julian, Michel Kalamarides, Matthias A. Karajannis, Bonnie Klein-Tasman, Pamela Knight, Bruce R. Korf, Shannon Langmead, Theresa LaVallee, Fawn Leigh, Donita Lightner, Carol Lin, Robert Listernick, Grant Liu, Mevo Marco, Carole L. Marcus, Gabriella Mariani, Staci Martin, Victor Mautner, Vanessa L. Merker, Michael J. Ferguson, Chris Moertel, Jill A. Morris, Katrina Morris, Kathryn N. North, Fabio P. Nunes, Roger J. Packer, Laura Papi, Allyson Parry, Neha Patel, Jonathan M. Payne, Karen Peluso, Sebastian Perreault, Scott Plotkin, Tina Young Poussaint, Nancy Ratner, Karlyne M. Reilly, Vincent M. Riccardi, Kent A. Robertson, Claas Röhl, Deborah Rukin Gold, La Rosa Salvatore, Laura Schaffner Gray, Elizabeth K. Schorry, Claire Semerjian, Monica Sheridan, Chie‐Schin Shih, Carolyn Sidor, William H. Slattery, Miriam J. Smith, Kathy Sommer, Marigo Stathis, Matthew R. Steensma, Anat Stemmer‐Rachamimov, David A. Stevenson, Kari Struemph, Lara Sullivan, Mary Anne Tamula, Mary Thomas, Heather Thompson, James H. Tonsgard, Nicole J. Ullrich, Sharad K. Verma, David Viskochil, Ana‐Maria Vranceanu, Ute Wahlländer, Karin S. Walsh, D. Bradley Welling, Ralph Wenzel, Trish Whitcomb, Brigitte C. Widemann, Victoria Williams, David S. Wolf, Pamela L. Wolters, Kaleb Yohay,

Glioma Diagnosis and Treatment

Neurofibromatosis type 1 (NF1) is associated with neurocognitive deficits that can impact everyday functioning of children, adolescents, and adults with this disease. However, there is little agreement regarding measures to use as cognitive endpoints in clinical trials. This article describes the work of the Neurocognitive Committee of the Response Evaluation in Neurofibromatosis and Schwannomatosis (REiNS) International Collaboration. The goal of this committee is to identify standardized and specific cognitive assessment tools for use in NF clinical trials. The committee first identified cognitive domains relevant to NF1 and prioritized attention as the first domain of focus given prior and current trends in NF1 cognitive clinical trials. Performance measures and behavioral rating questionnaires of attention were reviewed by the group using established criteria to assess patient characteristics, psychometric properties, and feasibility. The highest rated tests underwent side-by-side comparison. The Digit Span subtest from the Wechsler scales was given the highest ratings of the performance measures due to its good psychometrics, feasibility, utility across a wide age range, and extensive use in previous research. The Conners scales achieved the highest ratings of the behavioral questionnaires for similar reasons. Future articles will focus on other cognitive domains, with the ultimate goal of achieving agreement for cognitive endpoints that can be used across NF clinical trials.