Reasons for Failed Trials of Disease-Modifying Treatments for Alzheimer Disease and Their Contribution in Recent Research

Biomedicines - Tập 7 Số 4 - Trang 97
Konstantina G. Yiannopoulou1, Aikaterini Anastasiou2, Venetia Zachariou3, Sigliti‐Henrietta Pelidou4
1Memory Center, Neurological Department, Henry Dunant Hospital Center, 107 Mesogeion Avenue, 11526 Athens, Greece
2Medical School of Athens, National and Kapodistrian University of Athens, 11526 Athens, Greece
3Icahn School of Medicine at Mount Sinai, Nash family Department of Neurosciences, Department of Pharmacological Sciences, and Friedman Brain Institute, New York, NY 11004, USA
4Department of Neurology, University of Ioannina, University Hospital of Ioannina, 45500 Ioannina, Greece

Tóm tắt

Despite all scientific efforts and many protracted and expensive clinical trials, no new drug has been approved by FDA for treatment of Alzheimer disease (AD) since 2003. Indeed, more than 200 investigational programs have failed or have been abandoned in the last decade. The most probable explanations for failures of disease-modifying treatments (DMTs) for AD may include late initiation of treatments during the course of AD development, inappropriate drug dosages, erroneous selection of treatment targets, and mainly an inadequate understanding of the complex pathophysiology of AD, which may necessitate combination treatments rather than monotherapy. Clinical trials’ methodological issues have also been criticized. Drug-development research for AD is aimed to overcome these drawbacks. Preclinical and prodromal AD populations, as well as traditionally investigated populations representing all the clinical stages of AD, are included in recent trials. Systematic use of biomarkers in staging preclinical and prodromal AD and of a single primary outcome in trials of prodromal AD are regularly integrated. The application of amyloid, tau, and neurodegeneration biomarkers, including new biomarkers—such as Tau positron emission tomography, neurofilament light chain (blood and Cerebrospinal fluid (CSF) biomarker of axonal degeneration) and neurogranin (CSF biomarker of synaptic functioning)—to clinical trials allows more precise staging of AD. Additionally, use of Bayesian statistics, modifiable clinical trial designs, and clinical trial simulators enrich the trial methodology. Besides, combination therapy regimens are assessed in clinical trials. The above-mentioned diagnostic and statistical advances, which have been recently integrated in clinical trials, are relevant to the recent failures of studies of disease-modifying treatments. Their experiential rather than theoretical origins may better equip potentially successful drug-development strategies.

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Tài liệu tham khảo

Kingston, 2018, Forecasting the care needs of the older population in England over the next 20 years: Estimates from the Population Ageing and Care Simulation (PACSim) modelling study, Lancet Public Health., 3, e447, 10.1016/S2468-2667(18)30118-X

Scheltens, 2016, Alzheimer’s disease, Lancet, 388, 505, 10.1016/S0140-6736(15)01124-1

Cummings, 2019, Alzheimer’s disease drug development pipeline: 2019, Alzheimer Dement., 5, 272, 10.1016/j.trci.2019.05.008

Anderson, 2017, Why do so many clinical trials of therapies for Alzheimer’s disease fail?, Lancet, 25, 2327, 10.1016/S0140-6736(17)32399-1

Atri, 2019, Current and Future Treatments in Alzheimer’s Disease, Semin Neurol., 39, 227, 10.1055/s-0039-1678581

Gauthier, 2016, Why has therapy development for dementia failed in the last two decades?, Alzheimers Dement., 12, 60, 10.1016/j.jalz.2015.12.003

Cummings, 2019, Treatment Combinations for Alzheimer’s Disease: Current and Future Pharmacotherapy Options, J. Alzheimer Dis., 67, 779, 10.3233/JAD-180766

Dubois, 2016, Preclinical Alzheimer’s disease: Definition, natural history, and diagnostic criteria, Alzheimer Dement., 12, 292, 10.1016/j.jalz.2016.02.002

Yiannopoulou, 2013, Current and future treatments for Alzheimer’s disease, Ther. Adv. Neurol. Disord., 6, 19, 10.1177/1756285612461679

Anand, 2017, The present and future of pharmacotherapy of Alzheimer’s disease: A comprehensive review, Eur. J. Pharmacol., 815, 364, 10.1016/j.ejphar.2017.09.043

Bondi, 2017, Alzheimer’s Disease: Past, Present, and Future, J. Int. Neuropsychol Soc., 23, 818, 10.1017/S135561771700100X

Parasrampuria, 2018, Why Drugs Fail in Late Stages of Development: Case Study Analyses from the Last Decade and Recommendations, AAPS J., 20, 46, 10.1208/s12248-018-0204-y

Doody, 2013, Alzheimer’s Disease Cooperative Study Steering Committee, Siemers, E.; Sethuraman, G.; Mohs, R.; Semagacestat Study Group. A phase 3 trial of semagacestat for treatment of Alzheimer’s disease, N. Engl. J. Med., 369, 341, 10.1056/NEJMoa1210951

Coric, 2015, Targeting Prodromal Alzheimer Disease With Avagacestat: A Randomized Clinical Trial, JAMA Neurol., 72, 1324, 10.1001/jamaneurol.2015.0607

Muntimadugu, 2016, Intranasal delivery of nanoparticle encapsulated tarenflurbil: A potential brain targeting strategy for Alzheimer’s disease, Eur. J. Pharm. Sci., 20, 224, 10.1016/j.ejps.2016.05.012

Burki, 2018, Alzheimer’s disease research: The future of BACE inhibitors, Lancet, 23, 2486, 10.1016/S0140-6736(18)31425-9

Egan, 2019, Randomized Trial of Verubecestat for Prodromal Alzheimer’s Disease, N. Engl. J. Med., 11, 1408, 10.1056/NEJMoa1812840

Henley, 2019, Preliminary Results of a Trial of Atabecestat in Preclinical Alzheimer’s Disease, N. Engl. J. Med., 11, 1483, 10.1056/NEJMc1813435

Vandenberghe, 2016, Bapineuzumab 3000 and 3001 Clinical Study Investigators. Bapineuzumab for mild to moderate Alzheimer’s disease in two global, randomized, phase 3 trials, Alzheimers Res. Ther., 8, 18, 10.1186/s13195-016-0189-7

The Lancet Neurology (2017). Solanezumab: Too late in mild Alzheimer’s disease?. Lancet Neurol., 16, 97.

Scwarz, 2019, Magnetic resonance imaging measures of brain atrophy from the EXPEDITION3 trial in mild Alzheimer’s disease, Alzheimers Dement., 5, 328, 10.1016/j.trci.2019.05.007

Relkin, 2017, A phase 3 trial of IV immunoglobulin for Alzheimer disease, Neurology, 2, 1768, 10.1212/WNL.0000000000003904

Wilcock, 2018, Potential of Low Dose Leuco-Methylthioninium Bis(Hydromethanesulphonate)(LMTM) Monotherapy for Treatment of Mild Alzheimer’s Disease: Cohort Analysis as Modified Primary Outcome in a Phase III Clinical Trial, J. Alzheimers Dis., 61, 435, 10.3233/JAD-170560

Ower, 2018, Temporal association patterns and dynamics of amyloid-β and tau in Alzheimer’s disease, Eur. J. Epidemiol., 33, 657, 10.1007/s10654-017-0326-z

Tariot, 2019, The Alzheimer’s Prevention Initiative Generation Program: Study design of two randomized controlled trials for individuals at risk for clinical onset of Alzheimer’s disease, Alzheimers Dement., 12, 216

Wisniewski, 2015, Immunotherapeutic approaches for Alzheimer’s disease, Neuron, 85, 1162, 10.1016/j.neuron.2014.12.064

Xu, 2017, Synthesis and biological evaluation of deferiprone-resveratrol hybrids as antioxidants, Aβ1-42 aggregation inhibitors and metal-chelating agents for Alzheimer’s disease, Eur. J. Med. Chem., 15, 174, 10.1016/j.ejmech.2016.12.045

Villemagne, 2017, A randomized, exploratory molecular imaging study targeting amyloid β with a novel 8-OH quinoline in Alzheimer’s disease: The PBT2-204 IMAGINE study, Alzheimers Dement., 3, 622, 10.1016/j.trci.2017.10.001

Cho, 2014, ID1201, the ethanolic extract of the fruit of Melia toosendan ameliorates impairments in spatial learning and reduces levels of amyloid beta in 5XFAD mice, Neurosci. Lett., 583, 170, 10.1016/j.neulet.2014.09.036

Cummings, 2018, The National Institute on Aging-Alzheimer’s Association framework on Alzheimer’s disease: Application to clinical trials, Alzheimers Dement., 15, 172, 10.1016/j.jalz.2018.05.006

Jack, 2018, NIA-AA research framework: Toward a biological definition of Alzheimer’s disease, Alzheimers Dement., 14, 535, 10.1016/j.jalz.2018.02.018

Molinuevo, 2018, Current state of Alzheimer’s fluid biomarkers, Acta. Neuropathol., 136, 821, 10.1007/s00401-018-1932-x

Pontecorvo, 2017, 18-AV-1451-A05 investigators. Relationships between flortaucipir PET tau binding and amyloid burden, clinical diagnosis, age and cognition, Brain., 140, 748

Giau, V.V., Bagyinszky, E., and An, S.S.A. (2019). Potential Fluid Biomarkers for the Diagnosis of Mild Cognitive Impairment. Int. J. Mol. Sci., 20.

Hansson, 2006, Association between CSF biomarkers and incipient Alzheimer’s disease in patients with mild cognitive impairment: A follow-up study, Lancet Neurol., 5, 228, 10.1016/S1474-4422(06)70355-6

Isaac, 2011, Qualification opinion of novel methodologies in the predementia stage of Alzheimer’s disease: Cerebrospinal fluid related biomarkers for drugs affecting amyloid burden—Regulatory considerations by European Medicines Agency focusing in improving benefit/risk in regulatory trials, Eur. Neuropsychopharmacol., 21, 781, 10.1016/j.euroneuro.2011.08.003

Lewczuk, 2018, Cerebrospinal fluid and blood biomarkers for neurodegenerative dementias: An update of the Consensus of the Task Force on Biological Markers in Psychiatry of the World Federation of Societies of Biological Psychiatry, World J. Biol. Psychiatry, 19, 244, 10.1080/15622975.2017.1375556

Janelidze, 2016, CSF Aβ42/Aβ40 and Aβ42/ Aβ38 ratios: Better diagnostic markers of Alzheimer disease, Ann. Clin. Transl. Neurol., 3, 154, 10.1002/acn3.274

Shen, 2018, Increased plasma beta-secretase 1 may predict conversion to Alzheimer’s disease dementia in individuals with mild cognitive impairment, Biol. Psychiatry, 83, 447, 10.1016/j.biopsych.2017.02.007

Sotero, 2016, Alzheimer’s Disease Neuroimaging Initiative. Early role of vascular dysregulation on late-onset Alzheimer’s disease based on multifactorial data-driven analysis, Nat. Commun., 7, 11934, 10.1038/ncomms11934

Guo, 2013, Heart-type fatty acid binding protein and vascular endothelial growth factor: Cerebrospinal fluid biomarker candidates for Alzheimer’s disease, Eur. Arch. Psychiatry Clin. Neurosci., 263, 553, 10.1007/s00406-013-0405-4

Desikan, 2013, Heart fatty acid binding protein and Aβ-associated Alzheimer’s neurodegeneration, Mol. Neurodegener, 8, 39, 10.1186/1750-1326-8-39

Chiasserini, 2017, Differential role of CSF fatty acid binding protein 3, α-synuclein, and Alzheimer’s disease core biomarkers in Lewy body disorders and Alzheimer’s dementia, Alzheimers Res. Ther., 9, 52, 10.1186/s13195-017-0276-4

Gispert, 2016, Cerebrospinal fluid’s TREM2 levels are associated with gray matter volume increases and reduced diffusivity in early Alzheimer’s disease, Alzheimers Dement., 12, 1259, 10.1016/j.jalz.2016.06.005

Tan, 2017, Higher peripheral TREM2 mRNA levels relate to cognitive deficits and hippocampal atrophy in Alzheimer’s disease and amnestic mild cognitive impairment, J. Alzheimers Dis., 58, 413, 10.3233/JAD-161277

Pegueroles, 2017, YKL-40 (Chitinase 3-like I) is expressed in a subset of astrocytes in Alzheimer’s disease and other tauopathies, J. Neuroinfammation, 14, 118, 10.1186/s12974-017-0893-7

Tarawneh, 2016, Diagnostic and prognostic utility of the synaptic marker neurogranin in Alzheimer disease, JAMA Neurol., 73, 561, 10.1001/jamaneurol.2016.0086

Mavroudis, 2019, A meta-analysis on CSF neurogranin levels for the diagnosis of Alzheimer’s disease and mild cognitive impairment, Aging Clin. Exp. Res., 28, 1

Sutphen, 2018, Longitudinal decreases in multiple cerebrospinal fluid biomarkers of neuronal injury in symptomatic late onset Alzheimer’s disease, Alzheimers Dement., 14, 869, 10.1016/j.jalz.2018.01.012

Toledo, 2013, CSF alpha-synuclein improves diagnostic and prognostic performance of CSF tau and Abeta in Alzheimer’s disease, Acta Neuropathol., 126, 683, 10.1007/s00401-013-1148-z

Williams, 2017, Blood-based oligomeric and other protein variant biomarkers to facilitate presymptomatic diagnosis and staging of Alzheimer’s disease, J. Alzheimers Dis., 58, 23, 10.3233/JAD-161116

Ayton, 2017, Association of cerebrospinal fluid ferritin level with preclinical cognitive decline in APOE-ɛ4 carriers, JAMA Neurol., 74, 122, 10.1001/jamaneurol.2016.4406

Chang, 2014, The roles of biomarkers of oxidative stress and antioxidant in Alzheimer’s disease: A systematic review, BioMed Res. Int., 2014, 1

Baquero, 2017, Potential oxidative stress biomarkers of mild cognitive impairment due to Alzheimer disease, J. Neurol. Sci., 373, 295, 10.1016/j.jns.2017.01.020

Torres, 2011, Peripheral oxidative stress biomarkers in mild cognitive impairment and Alzheimer’s disease, J. Alzheimers Dis., 26, 59, 10.3233/JAD-2011-110284

Clark, 2002, Increase of brain oxidative stress in mild cognitive impairment: A possible predictor of Alzheimer disease, Arch. Neurol., 59, 972, 10.1001/archneur.59.6.972

Cuevas, 2019, Lipid peroxidation biomarkers correlation with medial temporal atrophy in early Alzheimer Disease, Neurochem. Int., 129, 104519, 10.1016/j.neuint.2019.104519

Moslemnezhad, 2016, Altered plasma marker of oxidative DNA damage and total antioxidant capacity in patients with Alzheimer’s disease, Caspian J. Intern. Med., 7, 88

Cervellati, 2014, Systemic oxidative stress and conversion to dementia of elderly patients with mild cognitive impairment, BioMed. Res. Int., 2014, 309507, 10.1155/2014/309507

Chico, 2013, Oxidative stress and APO E polymorphisms in Alzheimer’s disease and in mild cognitive impairment, Free Radic. Res., 47, 569, 10.3109/10715762.2013.804622

Tarawneh, 2012, CSF VILIP-1 predicts rates of cognitive decline in early Alzheimer disease, Neurology, 78, 709, 10.1212/WNL.0b013e318248e568

Zetterberg, 2016, Neurofilament Light: A Dynamic Cross-Disease Fluid Biomarker for Neurodegeneration, Neuron., 91, 1, 10.1016/j.neuron.2016.06.030

Zetterberg, 2016, Alzheimer’s Disease Neuroimaging Initiative. Association of cerebrospinal fluid neurofilament light concentration with Alzheimer disease progression, JAMA Neurol., 73, 60, 10.1001/jamaneurol.2015.3037

Mattsson, 2017, Association of Plasma Neurofilament Light with Neurodegeneration in Patients with Alzheimer Disease, JAMA Neurol., 74, 557, 10.1001/jamaneurol.2016.6117

Zhou, 2017, Alzheimer’s Disease Neuroimaging Initiative. Plasma neurofilament light chain levels in Alzheimer’s disease, Neurosci. Lett., 650, 60, 10.1016/j.neulet.2017.04.027

Yuan, 2017, Neurofilaments and neurofilament proteins in health and disease, Cold Spring Harb. Perspect. Biol., 9, a018309, 10.1101/cshperspect.a018309

Mak, 2017, Structural neuroimaging in preclinical dementia: From microstructural deficits and grey matter atrophy to macroscale connectomic changes, Ageing Res. Rev., 35, 250, 10.1016/j.arr.2016.10.001

Albert, 2018, Predicting progression from normal cognition to mild cognitive impairment for individuals at 5 years, Brain, 141, 877, 10.1093/brain/awx365

Yin, 2013, Brain imaging of mild cognitive impairment and Alzheimer’s disease, Neural Regen. Res., 8, 435

Rabinovici, 2019, Association of Amyloid Positron Emission Tomography with Subsequent Change in Clinical Management Among Medicare Beneficiaries with Mild Cognitive Impairment or Dementia, Jama, 321, 1286, 10.1001/jama.2019.2000

Brier, 2016, Tau and Ab imaging, CSR measures, and cognition in Alzheimer’s disease, Sci. Transl. Med., 8, 338ra66, 10.1126/scitranslmed.aaf2362

Johnson, 2016, Tau positron emission tomographic imaging in aging and early Alzheimer disease, Ann. Neurol., 79, 110, 10.1002/ana.24546

Zhang, 2018, Cromolyn Reduces Levels of the Alzheimer’s Disease-Associated Amyloid β-Protein by Promoting Microglial Phagocytosis, Sci. Rep., 8, 1144, 10.1038/s41598-018-19641-2

Boada, 2016, Treatment of Alzheimer disease using combination therapy with plasma exchange and haemapheresis with albumin and intravenous immunoglobulin: Rationale and treatment approach of the AMBAR (Alzheimer Management by Albumin Replacement) study, Neurologia, 31, 473, 10.1016/j.nrl.2014.02.003

Tomaszewski, 2016, Combination therapy of anti-tau and anti-amyloid drugs for disease modification in early-stage Alzheimer’s disease: Socio-economic considerations modeled on treatments for tuberculosis, HIV/AIDS and breast cancer, J. Prev. Alzheimers Dis., 3, 164

Evans, 2010, Clinical trial structures, J. Exp. Stroke Transl. Med., 3, 8, 10.6030/1939-067X-3.1.8

Bateman, 2017, The DIAN-TU Next Generation Alzheimer’s prevention trial: Adaptive design and disease progression model, Alzheimers Dement., 13, 8, 10.1016/j.jalz.2016.07.005

Ritchie, 2016, European Prevention of Alzheimer’s Dementia Consortium Development of interventions for the secondary prevention of Alzheimer’s dementia: The European Prevention of Alzheimer’s Dementia (EPAD) project, Lancet Psychiatry, 3, 179, 10.1016/S2215-0366(15)00454-X

Satlin, 2016, Design of a Bayesian adaptive phase 2 proof-of-concept trial for BAN2401, a putative disease-modifying monoclonal antibody for the treatment of Alzheimer’s disease, Alzheimers Dement., 2, 1, 10.1016/j.trci.2016.01.001

Selkoe, 2019, Alzheimer disease and aducanumab: Adjusting our approach, Nat. Rev. Neurol., 15, 365, 10.1038/s41582-019-0205-1

Howard, R., and Liu, K.Y. (2019). Questions EMERGE as Biogen claims aducanumab turnaround. Nat. Rev. Neurol.

Morris, 2018, Questions concerning the role of amyloid-β in the definition, aetiology and diagnosis of Alzheimer’s disease, Acta Neuropathol., 136, 663, 10.1007/s00401-018-1918-8

Panza, 2019, Are antibodies directed against amyloid-β (Aβ) oligomers the last call for the Aβ hypothesis of Alzheimer’s disease?, Immunotherapy, 11, 3, 10.2217/imt-2018-0119