bioRxiv Science⌕ Search

Biology subjects

Martinez de Lagran, M.

Publications and source records attributed to Martinez de Lagran, M..

2 recordsLinked to original sources

Deficits in neuronal architecture but not over-inhibition are main determinants of reduced neuronal network activity in a mouse model of overexpression of Dyrk1A

Abnormal dendritic arbors, dendritic spine "dysgenesis" and excitation inhibition imbalance are main traits assumed to underlie impaired cognition and behavioral adaptation in intellectual disability. However, how these modifications actually contribute to functional properties of neuronal networks, such as signal integration or storage capacity is unknown. Here, we used a mouse model overexpressing Dyrk1A (Dual-specificity tyrosine [Y]-regulated kinase), one of the most relevant Down syndrome (DS) candidate genes, to gather quantitative data regarding hippocampal neuronal deficits produced by the overexpression of Dyrk1A in mice (TgDyrk1A; TG). TG mice showed impaired hippocampal recognition memory, altered excitation-inhibition balance and deficits in hippocampal CA1 LTP. We also detected for the first time that deficits in dendritic arborization in TG CA1 pyramidal neurons are layer-specific, with a reduction in the width of the stratum radiatum, the postsynaptic target site of CA3 excitatory neurons, but not in the stratum lacunosum-moleculare, which receives temporo-ammonic projections. To interrogate about the functional impact of layer-specific TG dendritic deficits we developed tailored computational multicompartmental models. Computational modelling revealed that neuronal microarchitecture alterations in TG mice lead to deficits in storage capacity, altered the integration of inputs from entorhinal cortex and hippocampal CA3 region onto CA1 pyramidal cells, important for coding place and temporal context and on connectivity and activity dynamics, with impaired the ability to reach high {gamma} oscillations. Contrary to what is assumed in the field, the reduced network activity in TG is mainly contributed by the deficits in neuronal architecture and to a lesser extent by over-inhibition. Finally, given that therapies aimed at improving cognition have also been tested for their capability to recover dendritic spine deficits and excitation-inhibition imbalance, we also tested the short- and long-term changes produced by exposure to environmental enrichment (EE). Exposure to EE normalized the excitation inhibition imbalance and LTP, and had beneficial effects on short-term recognition memory. Importantly, it produced massive but transient dendritic remodeling of hippocampal CA1, that led to recovery of high {gamma} oscillations, the main readout of synchronization of CA1 neurons, in our simulations. However, those effects where not stable and were lost after EE discontinuation. We conclude that layer-specific neuromorphological disturbances produced by Dyrk1A overexpression impair coding place and temporal context. Our results also suggest that treatments targeting structural plasticity, such as EE, even though hold promise towards improved treatment of intellectual disabilities, only produce temporary recovery, due to transient dendritic remodeling.

neuroscience↗

The X-linked splicing regulator MBNL3 has been co-opted to restrict placental growth in eutherians

The eutherian placenta is a major site for parental genetic conflict. Here, we identify the X-linked Mbnl3 gene as a novel player in this dispute. Mbnl3 belongs to an RNA binding protein family whose members regulate alternative splicing and other aspects of RNA metabolism in association with cellular differentiation. We find that, in eutherians, Mbnl3 has become specifically expressed in placenta and has undergone accelerated sequence evolution leading to changes in its RNA binding specificities. Although its molecular roles are partly redundant with those of Mbnl2, Mbnl3 has also acquired novel biological functions. In particular, whereas Mbnl2;Mbnl3 double knockout mice display severe placental maturation defects leading to strong histological and functional abnormalities, Mbnl3 knockout alone results in increased placental growth and favors placental and fetal resource allocation during limiting conditions.

evolutionary biology↗