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Individualized atrophy scores predict dementia onset in familial frontotemporal lobar degeneration
- Staffaroni, Adam M;
- Cobigo, Yann;
- Goh, Sheng‐Yang M;
- Kornak, John;
- Bajorek, Lynn;
- Chiang, Kevin;
- Appleby, Brian;
- Bove, Jessica;
- Bordelon, Yvette;
- Brannelly, Patrick;
- Brushaber, Danielle;
- Caso, Christina;
- Coppola, Giovanni;
- Dever, Reilly;
- Dheel, Christina;
- Dickerson, Bradford C;
- Dickinson, Susan;
- Dominguez, Sophia;
- Domoto‐Reilly, Kimiko;
- Faber, Kelly;
- Ferrall, Jessica;
- Fields, Julie A;
- Fishman, Ann;
- Fong, Jamie;
- Foroud, Tatiana;
- Forsberg, Leah K;
- Gavrilova, Ralitza;
- Gearhart, Debra;
- Ghazanfari, Behnaz;
- Ghoshal, Nupur;
- Goldman, Jill;
- Graff‐Radford, Jonathan;
- Graff‐Radford, Neill;
- Grant, Ian;
- Grossman, Murray;
- Haley, Dana;
- Heuer, Hilary W;
- Hsiung, Ging‐Yuek;
- Huey, Edward D;
- Irwin, David J;
- Jones, David T;
- Jones, Lynne;
- Kantarci, Kejal;
- Karydas, Anna;
- Kaufer, Daniel I;
- Kerwin, Diana R;
- Knopman, David S;
- Kraft, Ruth;
- Kramer, Joel H;
- Kremers, Walter K;
- Kukull, Walter A;
- Litvan, Irene;
- Ljubenkov, Peter A;
- Lucente, Diane;
- Lungu, Codrin;
- Mackenzie, Ian R;
- Maldonado, Miranda;
- Manoochehri, Masood;
- McGinnis, Scott M;
- McKinley, Emily;
- Mendez, Mario F;
- Miller, Bruce L;
- Multani, Namita;
- Onyike, Chiadi;
- Padmanabhan, Jaya;
- Pantelyat, Alex;
- Pearlman, Rodney;
- Petrucelli, Len;
- Potter, Madeline;
- Rademakers, Rosa;
- Ramos, Eliana Marisa;
- Rankin, Katherine P;
- Rascovsky, Katya;
- Roberson, Erik D;
- Rogalski, Emily;
- Sengdy, Pheth;
- Shaw, Leslie M;
- Syrjanen, Jeremy;
- Tartaglia, M Carmela;
- Tatton, Nadine;
- Taylor, Joanne;
- Toga, Arthur;
- Trojanowski, John Q;
- Weintraub, Sandra;
- Wang, Ping;
- Wong, Bonnie;
- Wszolek, Zbigniew;
- Boxer, Adam L;
- Boeve, Brad F;
- Rosen, Howard J;
- consortium, on behalf of the ARTFL LEFFTDS
- et al.
Published Web Location
https://www.ncbi.nlm.nih.gov/pubmed/?term=Individualized+atrophy+scores+predict+dementia+onset+in+familial+frontotemporal+lobar+degeneration.No data is associated with this publication.
Abstract
Introduction
Some models of therapy for neurodegenerative diseases envision starting treatment before symptoms develop. Demonstrating that such treatments are effective requires accurate knowledge of when symptoms would have started without treatment. Familial frontotemporal lobar degeneration offers a unique opportunity to develop predictors of symptom onset.Methods
We created dementia risk scores in 268 familial frontotemporal lobar degeneration family members by entering covariate-adjusted standardized estimates of brain atrophy into a logistic regression to classify asymptomatic versus demented participants. The score's predictive value was tested in a separate group who were followed up longitudinally (stable vs. converted to dementia) using Cox proportional regressions with dementia risk score as the predictor.Results
Cross-validated logistic regression achieved good separation of asymptomatic versus demented (accuracy = 90%, SE = 0.06). Atrophy scores predicted conversion from asymptomatic or mildly/questionably symptomatic to dementia (HR = 1.51, 95% CI: [1.16,1.98]).Discussion
Individualized quantification of baseline brain atrophy is a promising predictor of progression in asymptomatic familial frontotemporal lobar degeneration mutation carriers.Many UC-authored scholarly publications are freely available on this site because of the UC's open access policies. Let us know how this access is important for you.