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@ARTICLE{Bateman:277740,
author = {Bateman, Randall J and Li, Yan and McDade, Eric M and
Llibre-Guerra, Jorge J and Clifford, David B and Atri,
Alireza and Mills, Susan L and Santacruz, Anna M and Wang,
Guoqiao and Supnet, Charlene and Benzinger, Tammie L S and
Gordon, Brian A and Ibanez, Laura and Klein, Gregory and
Baudler, Monika and Doody, Rachelle S and Delmar, Paul and
Kerchner, Geoffrey A and Bittner, Tobias and Wojtowicz,
Jakub and Bonni, Azad and Fontoura, Paulo and Hofmann,
Carsten and Kulic, Luka and Hassenstab, Jason and
Aschenbrenner, Andrew J and Perrin, Richard J and Cruchaga,
Carlos and Renton, Alan E and Xiong, Chengjie and Goate,
Alison A and Morris, John C and Holtzman, David M and
Snider, B Joy and Mummery, Catherine and Brooks, William S
and Wallon, David and Berman, Sarah B and Roberson, Erik and
Masters, Colin L and Galasko, Douglas R and Jayadev, Suman
and Sanchez-Valle, Rachel and Pariente, Jeremie and
Kinsella, Justin and van Dyck, Christopher H and Gauthier,
Serge and Hsiung, Ging-Yuek Robin and Masellis, Mario and
Dubois, Bruno and Honig, Lawrence S and Jack, Clifford R and
Daniels, Alisha and Aguillón, David and Allegri, Ricardo
and Chhatwal, Jasmeer and Day, Gregory and Fox, Nick C and
Huey, Edward and Ikeuchi, Takeshi and Jucker, Mathias and
Lee, Jae-Hong and Levey, Allan I and Levin, Johannes and
Lopera, Francisco and Roh, JeeHoon and Rosa-Neto, Pedro and
Schofield, Peter R},
collaboration = {Unit, Dominantly Inherited Alzheimer's Disease-Trials},
title = {{S}afety and efficacy of long-term gantenerumab treatment
in dominantly inherited {A}lzheimer's disease: an open-label
extension of the phase 2/3 multicentre, randomised,
double-blind, placebo-controlled platform {DIAN}-{TU}
trial.},
journal = {The lancet},
volume = {24},
number = {4},
issn = {1474-4422},
address = {London},
publisher = {Lancet Publ. Group},
reportid = {DZNE-2025-00461},
pages = {316 - 330},
year = {2025},
abstract = {Amyloid 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.},
keywords = {Humans / Antibodies, Monoclonal, Humanized: therapeutic use
/ Antibodies, Monoclonal, Humanized: pharmacology /
Antibodies, Monoclonal, Humanized: administration $\&$
dosage / Double-Blind Method / Male / Female / Alzheimer
Disease: drug therapy / Alzheimer Disease: genetics / Middle
Aged / Treatment Outcome / Adult / Aged / Antibodies,
Monoclonal, Humanized (NLM Chemicals) / gantenerumab (NLM
Chemicals) / solanezumab (NLM Chemicals)},
cin = {AG Jucker},
ddc = {610},
cid = {I:(DE-2719)1210001},
pnm = {352 - Disease Mechanisms (POF4-352)},
pid = {G:(DE-HGF)POF4-352},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:40120616},
doi = {10.1016/S1474-4422(25)00024-9},
url = {https://pub.dzne.de/record/277740},
}