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DOI 10.1016/j.jalz.2019.04.007

Медицинская библиотека · Alzheimer's & dementia : the journal of the Alzheimer's Association · 2020

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, ARTFL/LEFFTDS consortium

СтатьяПоддержка NIH: внешняя программаПоддержка исследования: вне правительства СШАПеревод пока не подготовлен

Аннотация

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.

Для цитирования: 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, ARTFL/LEFFTDS consortium. Individualized atrophy scores predict dementia onset in familial frontotemporal lobar degeneration.. Alzheimer's & dementia : the journal of the Alzheimer's Association. 2020. DOI: 10.1016/j.jalz.2019.04.007.

Предметные рубрики MeSH

AtrophyBrainC9orf72 ProteinFemaleFrontotemporal DementiaGenetic Predisposition to DiseaseHumansImage Processing, Computer-AssistedMagnetic Resonance ImagingMaleMiddle AgedMutationNeuropsychological TestsProgranulinstau Proteins
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