000277740 001__ 277740 000277740 005__ 20250420001112.0 000277740 0247_ $$2doi$$a10.1016/S1474-4422(25)00024-9 000277740 0247_ $$2pmid$$apmid:40120616 000277740 0247_ $$2ISSN$$a1474-4422 000277740 0247_ $$2ISSN$$a1474-4465 000277740 0247_ $$2altmetric$$aaltmetric:175328799 000277740 037__ $$aDZNE-2025-00461 000277740 041__ $$aEnglish 000277740 082__ $$a610 000277740 1001_ $$aBateman, Randall J$$b0 000277740 245__ $$aSafety and efficacy of long-term gantenerumab treatment in dominantly inherited Alzheimer's disease: an open-label extension of the phase 2/3 multicentre, randomised, double-blind, placebo-controlled platform DIAN-TU trial. 000277740 260__ $$aLondon$$bLancet Publ. Group$$c2025 000277740 3367_ $$2DRIVER$$aarticle 000277740 3367_ $$2DataCite$$aOutput Types/Journal article 000277740 3367_ $$0PUB:(DE-HGF)16$$2PUB:(DE-HGF)$$aJournal Article$$bjournal$$mjournal$$s1744705350_28087 000277740 3367_ $$2BibTeX$$aARTICLE 000277740 3367_ $$2ORCID$$aJOURNAL_ARTICLE 000277740 3367_ $$00$$2EndNote$$aJournal Article 000277740 520__ $$aAmyloid plaque removal by monoclonal antibody therapies slows clinical progression in symptomatic Alzheimer's disease; however, the potential for delaying the onset of clinical symptoms in asymptomatic people is unknown. The Dominantly Inherited Alzheimer Network Trials Unit (DIAN-TU) is an ongoing platform trial assessing the safety and efficacy of multiple investigational products in participants with dominantly inherited Alzheimer's disease (DIAD). Based on findings of amyloid removal and downstream biological effects from the gantenerumab group of the platform trial, we continued a 3-year open-label extension (OLE) study to assess the safety and efficacy of long-term treatment with high doses of gantenerumab.The randomised, placebo-controlled, double-blind, phase 2/3 multi-arm trial (DIAN-TU-001) assessed solanezumab or gantenerumab versus placebo in participants who were between 15 years before and 10 years after their estimated years to symptom onset and who had a Clinical Dementia Rating (CDR) global score of 0 (cognitively normal) to 1 (mild dementia). This study was followed by an OLE study of gantenerumab treatment, conducted at 18 study sites in Australia, Canada, France, Ireland, Puerto Rico, Spain, the UK, and the USA. For inclusion in the OLE, participants at risk for DIAD had participated in the double-blind period of DIAN-TU-001 and were required to know their mutation status. We investigated increasing doses of subcutaneous gantenerumab up to 1500 mg every 2 weeks. Due to the lack of a regulatory path for gantenerumab, the study was stopped early after a prespecified interim analysis (when most participants had completed 2 years of treatment) of the clinical measure CDR-Sum of Boxes (CDR-SB). The primary outcome for the final analysis was the amyloid plaque measure 11C-Pittsburgh compound-B positron emission tomography (PiB-PET) standardised uptake value ratio (SUVR [PiB-PET SUVR]) at 3 years, assessed in the modified intention-to-treat group (mITT; defined as participants who received any gantenerumab treatment post-OLE baseline, had at least one PiB-PET SUVR assessment before gantenerumab treatment, and a post-baseline assessment). All participants who received at least one dose of study drug in the OLE were included in the safety analysis. DIAN-TU-001 (NCT01760005) and the OLE (NCT06424236) are registered with ClinicalTrials.gov.Of 74 participants who were recruited into the OLE study between June 3, 2020, and April 22, 2021, 73 were enrolled and received gantenerumab treatment. 47 (64%) stopped dosing due to early termination of the study by the sponsor, and 13 (18%) prematurely discontinued the study for other reasons; 13 people completed 3 years of treatment. The mITT group for the primary analysis comprised 55 participants. At the interim analysis, the hazard ratio for clinical decline of CDR-SB in asymptomatic mutation carriers was 0·79 (n=53 [95% CI 0·47 to 1·32]) for participants who were treated with gantenerumab in either the double-blind or OLE period (Any Gant), and 0·53 (n=22 [0·27 to 1·03]) for participants who were treated with gantenerumab the longest (Longest Gant). At the final analysis, the adjusted mean change from OLE baseline to year 3 in PiB-PET SUVR was -0·71 SUVR (95% CI -0·88 to -0·53, p<0·0001). Amyloid-related imaging abnormalities occurred in 53% (39 of 73) of participants: 47% (34 of 73) with microhaemorrhages, 30% (22 of 73) with oedema, and 6% (five of 73) were associated with superficial siderosis. No treatment-associated macrohaemorrhages or deaths occurred.Partial or short-term amyloid removal did not show significant clinical effects. However, long-term full amyloid removal potentially delayed symptom onset and dementia progression. Our conclusions are limited due to the OLE design and use of external controls and need to be confirmed in long-term trials.National Institute on Aging, Alzheimer's Association, GHR Foundation, and F Hoffmann-La Roche/Genentech. 000277740 536__ $$0G:(DE-HGF)POF4-352$$a352 - Disease Mechanisms (POF4-352)$$cPOF4-352$$fPOF IV$$x0 000277740 588__ $$aDataset connected to CrossRef, PubMed, , Journals: pub.dzne.de 000277740 650_7 $$2NLM Chemicals$$aAntibodies, Monoclonal, Humanized 000277740 650_7 $$04DF060P933$$2NLM Chemicals$$agantenerumab 000277740 650_7 $$05D6PWO0333$$2NLM Chemicals$$asolanezumab 000277740 650_2 $$2MeSH$$aHumans 000277740 650_2 $$2MeSH$$aAntibodies, Monoclonal, Humanized: therapeutic use 000277740 650_2 $$2MeSH$$aAntibodies, Monoclonal, Humanized: pharmacology 000277740 650_2 $$2MeSH$$aAntibodies, Monoclonal, Humanized: administration & dosage 000277740 650_2 $$2MeSH$$aDouble-Blind Method 000277740 650_2 $$2MeSH$$aMale 000277740 650_2 $$2MeSH$$aFemale 000277740 650_2 $$2MeSH$$aAlzheimer Disease: drug therapy 000277740 650_2 $$2MeSH$$aAlzheimer Disease: genetics 000277740 650_2 $$2MeSH$$aMiddle Aged 000277740 650_2 $$2MeSH$$aTreatment Outcome 000277740 650_2 $$2MeSH$$aAdult 000277740 650_2 $$2MeSH$$aAged 000277740 7001_ $$aLi, Yan$$b1 000277740 7001_ $$aMcDade, Eric M$$b2 000277740 7001_ $$aLlibre-Guerra, Jorge J$$b3 000277740 7001_ $$aClifford, David B$$b4 000277740 7001_ $$aAtri, Alireza$$b5 000277740 7001_ $$aMills, Susan L$$b6 000277740 7001_ $$aSantacruz, Anna M$$b7 000277740 7001_ $$aWang, Guoqiao$$b8 000277740 7001_ $$aSupnet, Charlene$$b9 000277740 7001_ $$aBenzinger, Tammie L S$$b10 000277740 7001_ $$aGordon, Brian A$$b11 000277740 7001_ $$aIbanez, Laura$$b12 000277740 7001_ $$aKlein, Gregory$$b13 000277740 7001_ $$aBaudler, Monika$$b14 000277740 7001_ $$aDoody, Rachelle S$$b15 000277740 7001_ $$aDelmar, Paul$$b16 000277740 7001_ $$aKerchner, Geoffrey A$$b17 000277740 7001_ $$aBittner, Tobias$$b18 000277740 7001_ $$aWojtowicz, Jakub$$b19 000277740 7001_ $$aBonni, Azad$$b20 000277740 7001_ $$aFontoura, Paulo$$b21 000277740 7001_ $$aHofmann, Carsten$$b22 000277740 7001_ $$aKulic, Luka$$b23 000277740 7001_ $$aHassenstab, Jason$$b24 000277740 7001_ $$aAschenbrenner, Andrew J$$b25 000277740 7001_ $$aPerrin, Richard J$$b26 000277740 7001_ $$aCruchaga, Carlos$$b27 000277740 7001_ $$aRenton, Alan E$$b28 000277740 7001_ $$aXiong, Chengjie$$b29 000277740 7001_ $$aGoate, Alison A$$b30 000277740 7001_ $$aMorris, John C$$b31 000277740 7001_ $$aHoltzman, David M$$b32 000277740 7001_ $$aSnider, B Joy$$b33 000277740 7001_ $$aMummery, Catherine$$b34 000277740 7001_ $$aBrooks, William S$$b35 000277740 7001_ $$aWallon, David$$b36 000277740 7001_ $$aBerman, Sarah B$$b37 000277740 7001_ $$aRoberson, Erik$$b38 000277740 7001_ $$aMasters, Colin L$$b39 000277740 7001_ $$aGalasko, Douglas R$$b40 000277740 7001_ $$aJayadev, Suman$$b41 000277740 7001_ $$aSanchez-Valle, Rachel$$b42 000277740 7001_ $$aPariente, Jeremie$$b43 000277740 7001_ $$aKinsella, Justin$$b44 000277740 7001_ $$avan Dyck, Christopher H$$b45 000277740 7001_ $$aGauthier, Serge$$b46 000277740 7001_ $$aHsiung, Ging-Yuek Robin$$b47 000277740 7001_ $$aMasellis, Mario$$b48 000277740 7001_ $$aDubois, Bruno$$b49 000277740 7001_ $$aHonig, Lawrence S$$b50 000277740 7001_ $$aJack, Clifford R$$b51 000277740 7001_ $$aDaniels, Alisha$$b52 000277740 7001_ $$aAguillón, David$$b53 000277740 7001_ $$aAllegri, Ricardo$$b54 000277740 7001_ $$aChhatwal, Jasmeer$$b55 000277740 7001_ $$aDay, Gregory$$b56 000277740 7001_ $$aFox, Nick C$$b57 000277740 7001_ $$aHuey, Edward$$b58 000277740 7001_ $$aIkeuchi, Takeshi$$b59 000277740 7001_ $$0P:(DE-2719)2000010$$aJucker, Mathias$$b60$$udzne 000277740 7001_ $$0P:(DE-HGF)0$$aLee, Jae-Hong$$b61 000277740 7001_ $$aLevey, Allan I$$b62 000277740 7001_ $$0P:(DE-2719)2811659$$aLevin, Johannes$$b63$$udzne 000277740 7001_ $$aLopera, Francisco$$b64 000277740 7001_ $$aRoh, JeeHoon$$b65 000277740 7001_ $$aRosa-Neto, Pedro$$b66 000277740 7001_ $$aSchofield, Peter R$$b67 000277740 7001_ $$aUnit, Dominantly Inherited Alzheimer's Disease-Trials$$b68$$eCollaboration Author 000277740 773__ $$0PERI:(DE-600)2079704-7$$a10.1016/S1474-4422(25)00024-9$$gVol. 24, no. 4, p. 316 - 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