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DOI 10.1093/brain/awaf038

Медицинская библиотека · Brain : a journal of neurology · 2025

The landscape of autosomal-dominant Alzheimer's disease: global distribution and age of onset.

Liu Haiyan, Marsh Thomas W, Shi Xinyu, Renton Alan E, Bowling Kevin M, Ziegemeier Ellen, Wang Guoqiao, Cao Yuchen, Aristel Alisha, Li Jessie, Dickson Alexa, Perrin Richard J, Goate Alison M, Fernández Victoria, Day Gregory S, Doering Michelle, Daniels Alisha, Gordon Brian A, Benzinger Tammie L S, Hassenstab Jason, Ibanez Laura, Supnet-Bell Charlene, Xiong Chengjie, Allegri Ricardo, Berman Sarah B, Fox Nick C, Ryan Natalie S, Huey Edward D, Vöglein Jonathan, Noble James M, Roh Jee Hoon, Jucker Mathias, Laske Christoph, Ikeuchi Takeshi, Sanchez-Valle Raquel, Schofield Peter R, Mendez Patricio Chrem, Chhatwal Jasmeer P, Farlow Martin, Lee Jae-Hong, Levey Allan I, Levin Johannes, Lopera Francisco, Martins Ralph, Niimi Yoshiki, Rosa-Neto Pedro, Morris John C, Bateman Randall J, Karch Celeste M, Cruchaga Carlos, McDade Eric, Llibre-Guerra Jorge J

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Аннотация

We present a comprehensive global analysis of genetic variants associated with autosomal-dominant Alzheimer's disease (ADAD). A total of 550 variants in the APP, PSEN1 and PSEN2 genes were identified, of which 279 were classified as pathogenic or likely pathogenic based on American College of Medical Genetics and Genomics and the Association for Molecular Pathology criteria, utilizing data from the Dominantly Inherited Alzheimer Network (DIAN), literature and public databases. Symptomatic age at onset (AAO) data were estimated for 227 of these variants, allowing detailed characterization of their frequency, pathogenicity and AAO. Importantly, 226 variants met eligibility criteria for inclusion in disease-modifying clinical trials. Furthermore, we demonstrated the predictive value of mean variant AAO and parental AAO in predicting symptomatic AAO, validated against converters who became symptomatic during follow-up in the DIAN Observational Study. This dataset provides critical insights into the global landscape of ADAD and reveals the genetic and AAO heterogeneity of ADAD variants while refining variant trial eligibility criteria.

Для цитирования: Liu Haiyan, Marsh Thomas W, Shi Xinyu, Renton Alan E, Bowling Kevin M, Ziegemeier Ellen, Wang Guoqiao, Cao Yuchen, Aristel Alisha, Li Jessie, Dickson Alexa, Perrin Richard J, Goate Alison M, Fernández Victoria, Day Gregory S, Doering Michelle, Daniels Alisha, Gordon Brian A, Benzinger Tammie L S, Hassenstab Jason, Ibanez Laura, Supnet-Bell Charlene, Xiong Chengjie, Allegri Ricardo, Berman Sarah B, Fox Nick C, Ryan Natalie S, Huey Edward D, Vöglein Jonathan, Noble James M, Roh Jee Hoon, Jucker Mathias, Laske Christoph, Ikeuchi Takeshi, Sanchez-Valle Raquel, Schofield Peter R, Mendez Patricio Chrem, Chhatwal Jasmeer P, Farlow Martin, Lee Jae-Hong, Levey Allan I, Levin Johannes, Lopera Francisco, Martins Ralph, Niimi Yoshiki, Rosa-Neto Pedro, Morris John C, Bateman Randall J, Karch Celeste M, Cruchaga Carlos, McDade Eric, Llibre-Guerra Jorge J. The landscape of autosomal-dominant Alzheimer's disease: global distribution and age of onset.. Brain : a journal of neurology. 2025. DOI: 10.1093/brain/awaf038.

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