Literature DB >> 28119583

Distinct Parameters in the EEG of the PLP α-SYN Mouse Model for Multiple System Atrophy Reinforce Face Validity.

Lorenz Härtner1, Tobias W M Keil1, Matthias Kreuzer2, Eva Maria Fritz1, Gregor K Wenning3, Nadia Stefanova3, Thomas Fenzl1.   

Abstract

Multiple system atrophy (MSA) is a neurodegenerative movement disorder characterized by parkinsonian symptoms and cerebellar symptoms. Sleep disturbances also play a crucial role in MSA. One of the most convincing animal models in MSA research is the PLP α-SYN model, but to date no studies on sleep disturbances in this mouse model, frequently found in MSA patients are available. We identified spectral shifts within the EEG of the model, strikingly resembling results of clinical studies. We also characterized muscle activity during REM sleep, which is one of the key symptoms in REM sleep behavioral disorder. Spectral shifts and REM sleep-linked muscle activity were age dependent, supporting Face Validity of the PLP α-SYN model. We also strongly suggest our findings to be critically evaluated for Predictive Validity in future studies. Currently, research on MSA lacks potential compounds attenuating or curing MSA. Future drugs must prove its potential in animal models, for this our study provides potential biomarkers.

Entities:  

Keywords:  EEG; mouse model; multiple system atrophy; predictive validity; sleep

Year:  2017        PMID: 28119583      PMCID: PMC5222844          DOI: 10.3389/fnbeh.2016.00252

Source DB:  PubMed          Journal:  Front Behav Neurosci        ISSN: 1662-5153            Impact factor:   3.558


  70 in total

1.  Computation of measures of effect size for neuroscience data sets.

Authors:  Harald Hentschke; Maik C Stüttgen
Journal:  Eur J Neurosci       Date:  2011-11-14       Impact factor: 3.386

2.  Fully automated sleep deprivation in mice as a tool in sleep research.

Authors:  Thomas Fenzl; Christoph P N Romanowski; Cornelia Flachskamm; Karlheinz Honsberg; Erwin Boll; Arnold Hoehne; Mayumi Kimura
Journal:  J Neurosci Methods       Date:  2007-07-24       Impact factor: 2.390

3.  Hyperphosphorylation and insolubility of alpha-synuclein in transgenic mouse oligodendrocytes.

Authors:  Philipp J Kahle; Manuela Neumann; Laurence Ozmen; Veronika Muller; Helmut Jacobsen; Will Spooren; Babette Fuss; Barbara Mallon; Wendy B Macklin; Hideo Fujiwara; Masato Hasegawa; Takeshi Iwatsubo; Hans A Kretzschmar; Christian Haass
Journal:  EMBO Rep       Date:  2002-05-24       Impact factor: 8.807

4.  REM sleep behaviour disorder as the presenting symptom of multiple system atrophy.

Authors:  F Tison; G K Wenning; N P Quinn; S J Smith
Journal:  J Neurol Neurosurg Psychiatry       Date:  1995-03       Impact factor: 10.154

5.  Oxidative stress in transgenic mice with oligodendroglial alpha-synuclein overexpression replicates the characteristic neuropathology of multiple system atrophy.

Authors:  Nadia Stefanova; Markus Reindl; Manuela Neumann; Christian Haass; Werner Poewe; Philipp J Kahle; Gregor K Wenning
Journal:  Am J Pathol       Date:  2005-03       Impact factor: 4.307

6.  Sleep deprivation: effect on sleep stages and EEG power density in man.

Authors:  A A Borbély; F Baumann; D Brandeis; I Strauch; D Lehmann
Journal:  Electroencephalogr Clin Neurophysiol       Date:  1981-05

7.  Survival in multiple system atrophy.

Authors:  Anette Schrag; Gregor K Wenning; Niall Quinn; Yoav Ben-Shlomo
Journal:  Mov Disord       Date:  2008-01-30       Impact factor: 10.338

8.  Longitudinal change in REM sleep components in a patient with multiple system atrophy associated with REM sleep behavior disorder: paradoxical improvement of nocturnal behaviors in a progressive neurodegenerative disease.

Authors:  Naoko Tachibana; Yasunori Oka
Journal:  Sleep Med       Date:  2004-03       Impact factor: 3.492

Review 9.  Animal models of multiple system atrophy.

Authors:  Nadia Stefanova; Gregor K Wenning
Journal:  Clin Auton Res       Date:  2015-01-14       Impact factor: 4.435

Review 10.  Multiple system atrophy: a primary oligodendrogliopathy.

Authors:  Gregor K Wenning; Nadia Stefanova; Kurt A Jellinger; Werner Poewe; Michael G Schlossmacher
Journal:  Ann Neurol       Date:  2008-09       Impact factor: 10.422

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  8 in total

Review 1.  A Mouse Model of Multiple System Atrophy: Bench to Bedside.

Authors:  Nadia Stefanova
Journal:  Neurotherapeutics       Date:  2022-08-22       Impact factor: 6.088

Review 2.  Multiple system atrophy: experimental models and reality.

Authors:  Cassia Overk; Edward Rockenstein; Elvira Valera; Nadia Stefanova; Gregor Wenning; Eliezer Masliah
Journal:  Acta Neuropathol       Date:  2017-10-20       Impact factor: 17.088

3.  Progressive striatonigral degeneration in a transgenic mouse model of multiple system atrophy: translational implications for interventional therapies.

Authors:  Violetta Refolo; Francesco Bez; Alexia Polissidis; Daniela Kuzdas-Wood; Edith Sturm; Martina Kamaratou; Werner Poewe; Leonidas Stefanis; M Angela Cenci; Marina Romero-Ramos; Gregor K Wenning; Nadia Stefanova
Journal:  Acta Neuropathol Commun       Date:  2018-01-03       Impact factor: 7.801

4.  Assessment of the Retina of Plp-α-Syn Mice as a Model for Studying Synuclein-Dependent Diseases.

Authors:  Kathrin Kaehler; Hartwig Seitter; Adolf M Sandbichler; Bettina Tschugg; Gerald J Obermair; Nadia Stefanova; Alexandra Koschak
Journal:  Invest Ophthalmol Vis Sci       Date:  2020-06-03       Impact factor: 4.799

5.  Sleep/Wake Behavior and EEG Signatures of the TgF344-AD Rat Model at the Prodromal Stage.

Authors:  Matthias Kreuzer; Glenda L Keating; Thomas Fenzl; Lorenz Härtner; Christopher G Sinon; Ihab Hajjar; Vincent Ciavatta; David B Rye; Paul S García
Journal:  Int J Mol Sci       Date:  2020-12-05       Impact factor: 5.923

6.  Altered sleep behavior in a genetic mouse model of impaired fear extinction.

Authors:  Eva Maria Fritz; Matthias Kreuzer; Alp Altunkaya; Nicolas Singewald; Thomas Fenzl
Journal:  Sci Rep       Date:  2021-04-26       Impact factor: 4.379

Review 7.  A historical review of multiple system atrophy with a critical appraisal of cellular and animal models.

Authors:  David J Marmion; Wouter Peelaerts; Jeffrey H Kordower
Journal:  J Neural Transm (Vienna)       Date:  2021-10-06       Impact factor: 3.575

8.  Anle138b modulates α-synuclein oligomerization and prevents motor decline and neurodegeneration in a mouse model of multiple system atrophy.

Authors:  Antonio Heras-Garvin; Daniel Weckbecker; Sergey Ryazanov; Andrei Leonov; Christian Griesinger; Armin Giese; Gregor K Wenning; Nadia Stefanova
Journal:  Mov Disord       Date:  2018-11-19       Impact factor: 10.338

  8 in total

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