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Neuronal F-Box protein FBXO41 regulates synaptic transmission and hippocampal network maturation
Ana R A A Quadros1, Rocío Díez Arazola1, Andrea Romaguera Álvarez1
1Department of Functional Genomics, Vrije Universiteit (VU) Amsterdam, de Boelelaan 1085, 1081 HV Amsterdam, The Netherlands.
Iscience
|April 4, 2022
Summary
Loss of FBXO41, a neuron-specific E3 ligase, impairs synaptic transmission and postnatal brain development in mice. These deficits occur before observable behavioral changes in Fbxo41 knockout models.
Area of Science:
- Neuroscience
- Molecular Biology
- Developmental Biology
Background:
- FBXO41 is a neuron-specific E3 ligase subunit linked to epileptic encephalopathies.
- FBXO41 loss-of-function in mice (Fbxo41 KO) leads to behavioral deficits and early lethality.
Purpose of the Study:
- To investigate the role of FBXO41 in synaptic transmission and brain development.
- To characterize the cellular and circuit-level consequences of FBXO41 deficiency.
Main Methods:
- Single-cell electrophysiology in cultured neurons.
- Analysis of hippocampal slices from Fbxo41 KO mice.
- Two-photon calcium imaging of neuronal network activity.
Main Results:
- FBXO41 deficiency reduces synaptic vesicle release probability in excitatory neurons.
- Inhibitory neurons show decreased synaptophysin expression and impaired function.
- Fbxo41 KO mice exhibit smaller dentate gyrus, delayed neuronal migration, and altered network synchronicity.
Conclusions:
- FBXO41 is crucial for normal synaptic transmission and postnatal brain development.
- Defects in synaptic function and development precede behavioral deficits in Fbxo41 KO mice.
