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Wilke, J. B.

Publications and source records attributed to Wilke, J. B..

3 recordsLinked to original sources

AMPAR immunization induces progressive autoimmune encephalitis with autoreactive B cells in the brain

AMPA and NMDA receptors are central to synaptic plasticity and cognitive function. In anti-NMDA receptor and anti-AMPA receptor (AMPAR) encephalitis, autoantibodies targeting these receptors disrupt synaptic signaling, leading to severe neuropsychiatric symptoms. However, the cellular autoimmune responses and source of pathogenic autoantibodies during onset and progression of central nervous system (CNS) pathology remain poorly understood. By immunizing mice with intact AMPARs in proteoliposomes, we developed a mouse model of anti-AMPAR encephalitis. Mice developed rapidly progressing neuropsychiatric deficits, autoantibodies targeting the AMPAR amino-terminal domain (ATD) and IgG deposition in the brain, accompanied by reduced AMPAR detection. Throughout disease onset and progression, AMPAR-ATD-specific non-proliferating plasma cells and plasmablasts accumulated in the brain and were predominantly localized in AMPAR-expressing brain parenchyma. In contrast, differentiated AMPAR-ATD specific B cells were far less enriched in peripheral lymphoid tissues. Our results suggest that humoral autoimmune responses directly in the CNS drives disease progression in anti-AMPAR encephalitis.

neuroscience↗

Erythropoietin alleviates intellectual disability and autism-like behavior of mice caused by Zbtb20 haploinsufficiency, a construct-valid model of Primrose syndrome

Among the known genetic causes of syndromic autism spectrum disorders (ASD) are transcription factor deficiencies. In this regard, haploinsufficiency of the zinc finger and broad complex, tramtrack, bric and brac domain-containing protein 20 (ZBTB20) leads to a prototypical clinical picture, referred to as Primrose syndrome, comprising severe ASD symptoms together with intellectual disability. Here, we present a comprehensive behavioral and phenotypical characterization of Zbtb20+/- mice, a construct valid model of this thus far untreatable human condition. Zbtb20+/- mice exhibit diminished sociability, reduced vocalization, distinct repetitive behaviors, impaired cognitive flexibility, hyperactivity and hypoalgesia. Magnetic resonance imaging reveals increased volumes of hippocampus, cerebellum, brain matter, and whole brain, confirmed by postmortem brain weight measurements. Due to our previous observation of enhanced ZBTB20 expression in CA1 pyramidal neurons upon recombinant human erythropoietin (rhEPO) injections, we anticipated a mitigating effect through rhEPO treatment of Zbtb20 deficiency/Primrose syndrome. Indeed, after three weeks of alternate-day rhEPO injections, a remarkable improvement in the behavioral phenotype was observed. Our results highlight rhEPO as a first promising treatment for Primrose syndrome.

neuroscience↗

IntelliR: A comprehensive and standardized pipeline for automated profiling of higher cognition in mice

In the rapidly evolving field of rodent behavior research, observer-independent methods facilitate data collection within a social, stress-reduced, and thus more natural environment. A prevalent system in this research area is the IntelliCage, which empowers experimenters to design individual tasks and higher cognitive challenges for mice, driven by their motivation to access reward. The extensive amount and diversity of data provided by the IntelliCage system explains the growing demand for automated analysis among users. Here, we introduce IntelliR, a standardized pipeline for analyzing raw data generated by the IntelliCage software, as well as novel parameters including the cognition index, which enables comparison of performance across various challenges. With IntelliR, we provide the tools to implement and automatically analyze 3 challenges that we designed, encompassing spatial, episodic-like, and working memory with their respective reversal tests. Using results from 3 independent control cohorts of adult female wildtype mice, we demonstrate their ability to comprehend and learn the tasks, thereby improving their proficiency over time. To validate the sensitivity of our approach for detecting cognitive impairment, we used adult female NexCreERT2xRosa26-eGFP-DTA mice after tamoxifen induced diphtheria toxin-mediated ablation of pyramidal neurons in cortex and hippocampus. We observed deterioration in learning capabilities and cognition index across several tests. IntelliR can be readily integrated into and adapted for individual research, thereby improving time management and reproducibility of data analysis. HIGHLIGHTSO_LIIntelliR is a standardized pipeline for analyzing raw data of IntelliCage software. C_LIO_LIDomains include spatial, episodic-like, and working memory with reversals. C_LIO_LIWT mice (3 cohorts) comprehend, learn and improve proficiency over time. C_LIO_LICognition index permits comparison of performance across cognitive domains. C_LIO_LIMice with ablation of pyramidal neurons decline mainly in working memory. C_LI

animal behavior and cognition↗