Publications

Hope Center member publications

List of publications for the week of November 3, 2021

TREM2 modulates differential deposition of modified and non-modified Aβ species in extracellular plaques and intraneuronal deposits” (2021) Acta Neuropathologica Communications

TREM2 modulates differential deposition of modified and non-modified Aβ species in extracellular plaques and intraneuronal deposits
(2021) Acta Neuropathologica Communications, 9 (1), art. no. 168, . 

Joshi, P.a , Riffel, F.a , Kumar, S.a , Villacampa, N.b c , Theil, S.a , Parhizkar, S.d , Haass, C.d e f , Colonna, M.g , Heneka, M.T.b c , Arzberger, T.f h i , Herms, J.e f h , Walter, J.a

a Department of Neurology, University of Bonn, Venusberg-Campus 1, (Formerly Sigmund-Freud-Str. 25), Bonn, 53127, Germany
b Department of Neurodegenerative Diseases and Gerontopsychiatry, University Hospital Bonn, Bonn, Germany
c Neuroinflammation Unit, German Center for Neurodegenerative Diseases e. V. (DZNE), Bonn, Germany
d Biomedical Center (BMC), Faculty of Medicine, Ludwig-Maximilians-Universität München, Munich, Germany
e Munich Cluster for Systems Neurology (SyNergy), Munich, Germany
f Molecular Neurodegeneration Unit, German Center for Neurodegenerative Diseases e.V. (DZNE) Munich, Munich, Germany
g Department of Pathology and Immunology, Washington University School of Medicine, St. Louis, United States
h Center for Neuropathology and Prion Research, Ludwig-Maximilians-Universität München, Munich, Germany
i Department of Psychiatry and Psychotherapy, Ludwig-Maximilians-Universität München, Munich, Germany

Abstract
Progressive accumulation of Amyloid-β (Aβ) deposits in the brain is a characteristic neuropathological hallmark of Alzheimer’s disease (AD). During disease progression, extracellular Aβ plaques undergo specific changes in their composition by the sequential deposition of different modified Aβ species. Microglia are implicated in the restriction of amyloid deposits and play a major role in internalization and degradation of Aβ. Recent studies showed that rare variants of the Triggering Receptor Expressed on Myeloid cells 2 (TREM2) are associated with an increased risk for AD. Post-translational modifications of Aβ could modulate the interaction with TREM2, and the uptake by microglia. Here, we demonstrate that genetic deletion of TREM2 or expression of a disease associated TREM2 variant in mice lead to differential accumulation of modified and non-modified Aβ species in extracellular plaques and intraneuronal deposits. Human brains with rare TREM2 AD risk variants also showed altered deposition of modified Aβ species in the different brain lesions as compared to cases with the common variant of TREM2. These findings indicate that TREM2 plays a critical role in the development and the composition of Aβ deposits, not only in extracellular plaques, but also intraneuronally, that both could contribute to the pathogenesis of AD. © 2021, The Author(s).

Author Keywords
Aβ;  Intraneuronal;  Microglia;  Post-translational modification;  TREM2;  Vascular deposits

Funding details
115976
Deutsche ForschungsgemeinschaftDFGWA1477/6-6
Rheinische Friedrich-Wilhelms-Universität BonnUni Bonn388169927
Innovative Medicines InitiativeIMI

Document Type: Article
Publication Stage: Final
Source: Scopus

Sensory Percepts Elicited by Chronic Macro-Sieve Electrode Stimulation of the Rat Sciatic Nerve” (2021) Frontiers in Neuroscience

Sensory Percepts Elicited by Chronic Macro-Sieve Electrode Stimulation of the Rat Sciatic Nerve
(2021) Frontiers in Neuroscience, 15, art. no. 758427, . 

Chandra, N.S.a , McCarron, W.M.b , Yan, Y.b , Ruiz, L.C.a , Sallinger, E.G.c , Birenbaum, N.K.a , Burton, H.d , Green, L.e , Moran, D.W.a , Ray, W.Z.b , MacEwan, M.R.b

a Department of Biomedical Engineering, Washington University in St. Louis, St. Louis, MO, United States
b Department of Neurosurgery, Washington University School of Medicine in St. Louis, St. Louis, MO, United States
c Department of Biology, Washington University in St. Louis, St. Louis, MO, United States
d Department of Neuroscience, Washington University School of Medicine in St. Louis, St. Louis, MO, United States
e Department of Psychological Brain Sciences, Washington University in St. Louis, St. Louis, MO, United States

Abstract
Objective: Intuitive control of conventional prostheses is hampered by their inability to provide the real-time tactile and proprioceptive feedback of natural sensory pathways. The macro-sieve electrode (MSE) is a candidate interface to amputees’ truncated peripheral nerves for introducing sensory feedback from external sensors to facilitate prosthetic control. Its unique geometry enables selective control of the complete nerve cross-section by current steering. Unlike previously studied interfaces that target intact nerve, the MSE’s implantation requires transection and subsequent regeneration of the target nerve. Therefore, a key determinant of the MSE’s suitability for this task is whether it can elicit sensory percepts at low current levels in the face of altered morphology and caliber distribution inherent to axon regeneration. The present in vivo study describes a combined rat sciatic nerve and behavioral model developed to answer this question. Approach: Rats learned a go/no-go detection task using auditory stimuli and then underwent surgery to implant the MSE in the sciatic nerve. After healing, they were trained with monopolar electrical stimuli with one multi-channel and eight single-channel stimulus configurations. Psychometric curves derived by the method of constant stimuli (MCS) were used to calculate 50% detection thresholds and associated psychometric slopes. Thresholds and slopes were calculated at two time points 3 weeks apart. Main Results: For the multi-channel stimulus configuration, the average current required for stimulus detection was 19.37 μA (3.87 nC) per channel. Single-channel thresholds for leads located near the nerve’s center were, on average, half those of leads located near the periphery (54.92 μA vs. 110.71 μA, or 10.98 nC vs. 22.14 nC). Longitudinally, 3 of 5 leads’ thresholds decreased or remained stable over the 3-week span. The remaining two leads’ thresholds increased by 70–74%, possibly due to scarring or device failure. Significance: This work represents an important first step in establishing the MSE’s viability as a sensory feedback interface. It further lays the groundwork for future experiments that will extend this model to the study of other devices, stimulus parameters, and task paradigms. © Copyright © 2021 Chandra, McCarron, Yan, Ruiz, Sallinger, Birenbaum, Burton, Green, Moran, Ray and MacEwan.

Author Keywords
macro-sieve electrode;  nerve regeneration;  peripheral nerve stimulation;  rat behavior;  regenerative electrode;  sciatic nerve;  sensorimotor restoration;  sensory feedback

Funding details
Washington University in St. LouisWUSTL2017-WR
Hope Center for Neurological Disorders

Document Type: Article
Publication Stage: Final
Source: Scopus

Reimagining pilocytic astrocytomas in the context of pediatric low-grade gliomas” (2021) Neuro-Oncology

Reimagining pilocytic astrocytomas in the context of pediatric low-grade gliomas
(2021) Neuro-Oncology, 23 (10), pp. 1634-1646. 

Milde, T.a b c , Rodriguez, F.J.d , Barnholtz-Sloan, J.S.e f g , Patil, N.f g , Eberhart, C.G.d , Gutmann, D.H.h

a Hopp Children’s Cancer Center (KiTZ), Heidelberg, Germany
b Clinical Cooperation Unit Pediatric Oncology, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany
c Department of Pediatric Hematology and Oncology, Heidelberg University Hospital, Heidelberg, Germany
d Department of Pathology, Johns Hopkins University School of Medicine, Baltimore, MD, United States
e Department of Population and Quantitative Health Sciences, Case Western Reserve School of Medicine, Cleveland, OH, United States
f University Hospitals, Cleveland, OH, United States
g Central Brain Tumor Registry of the United States (CBTRUS), Hinsdale, IL, United States
h Department of Neurology, Washington University School of Medicine, St. Louis, MO, United States

Abstract
Pediatric low-grade gliomas (pLGGs) are the most common brain tumor in children and are associated with lifelong clinical morbidity. Relative to their high-grade adult counterparts or other malignant childhood brain tumors, there is a paucity of authenticated preclinical models for these pLGGs and an incomplete understanding of their molecular and cellular pathogenesis. While large-scale genomic profiling efforts have identified the majority of pathogenic driver mutations, which converge on the MAPK/ERK signaling pathway, it is now appreciated that these events may not be sufficient by themselves for gliomagenesis and clinical progression. In light of the recent World Health Organization reclassification of pLGGs, and pilocytic astrocytoma (PA), in particular, we review our current understanding of these pediatric brain tumors, provide a conceptual framework for future mechanistic studies, and outline the challenges and pressing needs for the pLGG clinical and research communities. © 2021 The Author(s) 2021. Published by Oxford University Press on behalf of the Society for Neuro-Oncology. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.

Author Keywords
BRAF;  cellular senescence;  low-grade glioma;  MEK;  neurofibromatosis type 1;  pediatric brain tumor;  pilocytic astrocytoma;  tumor microenvironment

Document Type: Article
Publication Stage: Final
Source: Scopus

Pathology Features of Immune and Inflammatory Myopathies, Including a Polymyositis Pattern, Relate Strongly to Serum Autoantibodies” (2021) Journal of Neuropathology and Experimental Neurology

Pathology Features of Immune and Inflammatory Myopathies, Including a Polymyositis Pattern, Relate Strongly to Serum Autoantibodies
(2021) Journal of Neuropathology and Experimental Neurology, 80 (9), pp. 812-820. 

Pestronk, A.a b , Choksi, R.a

a Department of Neurology, Washington University School of Medicine, St. Louis, MO, United States
b Department of Pathology and Immunology, Washington University School of Medicine, St. Louis, MO, United States

Abstract
We asked whether myopathology features of immune or inflammatory myopathies (IIM), without reference to clinical or laboratory attributes, correlate with serum autoantibodies. Retrospective study included 148 muscle biopsies with: B-cell inflammatory foci (BIM), myovasculopathy, perimysial pathology (IMPP), myofiber necrosis without perimysial or vessel damage or inflammation (MNec), inflammation and myofiber vacuoles or mitochondrial pathology (IM-VAMP), granulomas, chronic graft-versus-host disease, or none of these criteria. 18 IIM-related serum autoantibodies were tested. Strong associations between myopathology and autoantibodies included: BIM with PM/Scl-100 (63%; odds ratio [OR] = 72); myovasculopathies with TIF1-γ or NXP2 (70%; OR = 72); IMPP with Jo-1 (33%; OR = 28); MNec with SRP54 (23%; OR = 37); IM-VAMP with NT5C1a (95%; OR = 83). Hydroxymethylglutaryl-CoA reductase (HMGCR) antibodies related to presence of myofiber necrosis across all groups (82%; OR = 9), but not to one IIM pathology group. Our results validate characterizations of IIM by myopathology features, showing strong associations with some serum autoantibodies, another objective IIM-related marker. BIM with PM/Scl-100 antibodies can be described pathologically as polymyositis. Tif1-γ and NXP2 antibodies are both common in myovasculopathies. HMGCR antibodies associate with myofiber necrosis, but not one IIM pathology subtype. Relative association strengths of IIM-related autoantibodies to IIM myopathology features versus clinical characteristics require further study. © 2021 American Association of Neuropathologists, Inc. All rights reserved.

Author Keywords
Autoantibodies;  B-cells;  Immune;  Inflammatory;  Myopathy;  Myositis;  Necrosis

Document Type: Article
Publication Stage: Final
Source: Scopus

Apolipoprotein E ɛ4–related effects on cognition are limited to the Alzheimer’s disease spectrum” (2021) GeroScience

Apolipoprotein E ɛ4–related effects on cognition are limited to the Alzheimer’s disease spectrum
(2021) GeroScience, . 

Fernández, A.a b , Vaquero, L.a b , Bajo, R.c d , Zuluaga, P.c , Weiner, M.W.e , Saykin, A.J.f , Trojanowski, J.Q.g , Shaw, L.h , Toga, A.W.i , Beckett, L.j , Jack, C.R.k , Aisen, P.l , Petersen, R.C.k , Morris, J.C.m , Jagust, W.n , ADNI-Alzheimer’s Disease Neuroimaging Initiativeo

a Legal Medicine, Psychiatry, and Pathology Department, Faculty of Medicine, Complutense University of Madrid, Pza. Ramón Y Cajal, s/n, Ciudad Universitaria, Madrid, 28040, Spain
b Laboratory of Cognitive & Computational Neuroscience, Complutense and Polytechnic Universities of Madrid Joint Laboratory, Centre for Biomedical Technology, Pozuelo de Alarcón, Spain
c Statistics & Operations Research Department, Faculty of Medicine, Complutense University of Madrid, Madrid, Spain
d Electrical Engineering & Bioengineering Group (EE&B), Industrial Engineering Department, University of La Laguna, Tenerife, Spain
e University of California San Francisco—School of Medicine, Northern California Institute for research and education, USCF—VA Medical Center, 4150 Clement St., San Fancisco, CA 94,121, United States
f Center for Neuroimaging, School of Medicine, Indiana University, RADY, Indianapolis, IN GH4324, United States
g Center for Neurodegenerative Disease Research, School of Medicine, University of Pennsylvania, 3600 Spruce Street, 3d floor Maloney Building, Philadelphia, PA 10,104–4283, United States
h Hospital of the University of Pennsylvania, Pathology and Laboratory Medicine Dept, 7.103 Founders Pavilion, 3400 Spruce st., Philadelphia, PA 19104, United States
i USC Institute for Neuroimaging and Neuroinformatics, Keck School of Medicine—University of Southern California, SHN 2025 Zonal Av., Health Sciences Campus, Los Angeles, CA 90,033, United States
j Department of Public Health Sciences—University of California, Medical Sciences 1-C, One-Shield’s Ave., Davis, CA 95,616, United States
k Mayo’s Alzheimer’s Disease Research Center—Mayo Clinic, 200 First St. SW, Rochester, MN 55,905, United States
l Alzheimer’s Therapeutic Research Institute—Keck School of Medicine, University of Southern California, ATRI 9860 Mesa Rim Road, Health Sciences Campus, San Diego, CA 92,121, United States
m Center for Advanced Medicine—Memory Diagnostic Center, Washington University, 4921 Parkview Place, St Louis, MO 63,110, United States
n Helen Wills Neuroscience Institute, University of California, 132 Barker Hall, MC#3190, Berkeley, CA 94,720, United States

Abstract
Whether the deleterious effects of APOE4 are restricted to the Alzheimer’s disease (AD) spectrum or cause cognitive impairment irrespectively of the development of AD is still a matter of debate, and the focus of this study. Our analyses included APOE4 genotype, neuropsychological variables, amyloid-βeta (Aβ) and Tau markers, FDG-PET values, and hippocampal volumetry data derived from the healthy controls sample of the ADNI database. We formed 4 groups of equal size (n = 30) based on APOE4 carriage and amyloid-PET status. Baseline and follow-up (i.e., 48 months post-baseline) results indicated that Aβ-positivity was the most important factor to explain poorer cognitive performance, while APOE4 only exerted a significant effect in Aβ-positive subjects. Additionally, multiple regression analyses evidenced that, within the Aβ-positive sample, hippocampal volumetry explained most of the variability in cognitive performance for APOE4 carriers. These findings represent a strong support for the so-called preclinical/prodromal hypothesis, which states that the reported differences in cognitive performance between healthy carriers and non-carriers are mainly due to the APOE4’s capability to increase the risk of AD. Moreover, our results reinforce the notion that a synergistic interaction of Aβ and APOE4 elicits a neurodegenerative process in the hippocampus that might be the main cause of impaired cognitive performance. © 2021, The Author(s).

Author Keywords
Amyloid markers;  ApoE4;  Cognitive deterioration;  Cognitive phenotype;  Healthy aging;  Preclinical and prodromal Alzheimer’s disease

Funding details
National Institutes of HealthNIHU01 AG024904
U.S. Department of DefenseDODW81XWH-12-2-0012
National Institute on AgingNIA
National Institute of Biomedical Imaging and BioengineeringNIBIB
Alzheimer’s Disease Neuroimaging InitiativeADNI

Document Type: Article
Publication Stage: Article in Press
Source: Scopus

Pain induces adaptations in ventral tegmental area dopamine neurons to drive anhedonia-like behavior” (2021) Nature Neuroscience

Pain induces adaptations in ventral tegmental area dopamine neurons to drive anhedonia-like behavior
(2021) Nature Neuroscience, . 

Markovic, T.a b c d , Pedersen, C.E.e f , Massaly, N.a b c , Vachez, Y.M.a b c , Ruyle, B.a b c , Murphy, C.A.a , Abiraman, K.a , Shin, J.H.g , Garcia, J.J.c , Yoon, H.J.a b c , Alvarez, V.A.g , Bruchas, M.R.e f , Creed, M.C.a b c d h i , Morón, J.A.a b c d h

a Department of Anesthesiology, Washington University in St. Louis, St. Louis, MO, United States
b Pain Center, Washington University in St. Louis, St. Louis, MO, United States
c School of Medicine, Washington University in St. Louis, St. Louis, MO, United States
d Department of Neuroscience, Washington University in St. Louis, St. Louis, MO, United States
e Center for the Neurobiology of Addiction, Pain and Emotion, Departments of Anesthesiology and Pharmacology, University of Washington, Seattle, WA, United States
f Department of Bioengineering, University of Washington, Seattle, WA, United States
g Laboratory on Neurobiology of Compulsive Behaviors, National Institute on Alcohol Abuse and Alcoholism, Center on Compulsive Behaviors, Intramural Research Program, NIH, Bethesda, MD, United States
h Department of Psychiatry, Washington University in St. Louis, St. Louis, MO, United States
i Department of Biomedical Engineering, Washington University in St. Louis, St. Louis, MO, United States

Abstract
The persistence of negative affect in pain leads to co-morbid symptoms such as anhedonia and depression—major health issues in the United States. The neuronal circuitry and contribution of specific cellular populations underlying these behavioral adaptations remains unknown. A common characteristic of negative affect is a decrease in motivation to initiate and complete goal-directed behavior, known as anhedonia. We report that in rodents, inflammatory pain decreased the activity of ventral tegmental area (VTA) dopamine (DA) neurons, which are critical mediators of motivational states. Pain increased rostromedial tegmental nucleus inhibitory tone onto VTA DA neurons, making them less excitable. Furthermore, the decreased activity of DA neurons was associated with reduced motivation for natural rewards, consistent with anhedonia-like behavior. Selective activation of VTA DA neurons was sufficient to restore baseline motivation and hedonic responses to natural rewards. These findings reveal pain-induced adaptations within VTA DA neurons that underlie anhedonia-like behavior. © 2021, The Author(s), under exclusive licence to Springer Nature America, Inc.

Funding details
R01-DA049924
National Institutes of HealthNIHDA041781, DA041883, DA042499, DA042581, DA045463
National Institute on Drug AbuseNIDAR21-DA047127
Brain and Behavior Research FoundationBBRF27197
Whitehall Foundation2017-12-54
Israel National Road Safety AuthorityNRSAF31DA051124
National Alliance for Research on Schizophrenia and DepressionNARSAD

Document Type: Article
Publication Stage: Article in Press
Source: Scopus