eprintid: 10091061 rev_number: 30 eprint_status: archive userid: 608 dir: disk0/10/09/10/61 datestamp: 2020-02-13 11:57:42 lastmod: 2021-10-05 00:47:46 status_changed: 2020-02-13 11:57:42 type: article metadata_visibility: show creators_name: Curtis, D creators_name: Bakaya, K creators_name: Sharma, L creators_name: Bandyopadhyay, S title: Weighted burden analysis of exome‐sequenced late‐onset Alzheimer's cases and controls provides further evidence for a role for PSEN1 and suggests involvement of the PI3K/Akt/GSK‐3β and WNT signalling pathways ispublished: pub divisions: UCL divisions: B02 divisions: C08 divisions: D09 divisions: F99 keywords: C1R, LOAD, NDRG2, PIK3R1, PSEN1, TIAF1, tyrosine phosphatase note: This version is the author accepted manuscript. For information on re-use, please refer to the publisher’s terms and conditions. abstract: Previous studies have implicated common and rare genetic variants as risk factors for late‐onset Alzheimer's disease (LOAD). Here, weighted burden analysis was applied to over 10,000 exome‐sequenced subjects from the Alzheimer's Disease Sequencing Project. Analyses were carried out to investigate whether rare variants predicted to have a functional effect within a gene were more commonly seen in cases or in controls. Confirmatory results were obtained for TREM2, ABCA7, and SORL1. Additional support was provided for PSEN1 (p = 0.0002), which previously had been only weakly implicated in LOAD. There was suggestive evidence that functional variants in PIK3R1, WNT7A, C1R, and EXOC5 might increase risk and that variants in TIAF1 and/or NDRG2 might have a protective effect. Overall, there was strong evidence (p = 5 × 10−6) that variants in tyrosine phosphatase genes reduce the risk of developing LOAD. Because PIK3R1 variants are expected to impair PI3K/Akt/GSK‐3β signalling while variants in tyrosine phosphatase genes would enhance it, these findings are in line with those from animal models, suggesting that this pathway is protective against Alzheimer's disease. date: 2020-05 date_type: published official_url: https://doi.org/10.1111/ahg.12375 oa_status: green full_text_type: other language: eng primo: open primo_central: open_green verified: verified_manual elements_id: 1749450 doi: 10.1111/ahg.12375 lyricists_name: Curtis, David lyricists_id: DCURT26 actors_name: Austen, Jennifer actors_id: JAUST66 actors_role: owner full_text_status: public publication: Annals of Human Genetics volume: 84 number: 3 pagerange: 291-302 event_location: England citation: Curtis, D; Bakaya, K; Sharma, L; Bandyopadhyay, S; (2020) Weighted burden analysis of exome‐sequenced late‐onset Alzheimer's cases and controls provides further evidence for a role for PSEN1 and suggests involvement of the PI3K/Akt/GSK‐3β and WNT signalling pathways. Annals of Human Genetics , 84 (3) pp. 291-302. 10.1111/ahg.12375 <https://doi.org/10.1111/ahg.12375>. Green open access document_url: https://discovery.ucl.ac.uk/id/eprint/10091061/1/Andres_SAPER%20theory%20and%20practise%20-%20Final%20Accepted%20Version.pdf