Loss of Fmr1 causes subnucleus-specific decreases in dendritic complexity in mediodorsal thalamus

In the mediodorsal thalamus (MD), neurons of the medial (MD-M) and lateral (MD-L) subnuclei project to the medial prefrontal cortex (mPFC) to control complementary aspects of behavior, with MD-M supporting more of the social/emotional domain and MD-L promoting cognitive functions. Our recent work shows that MD-M and MD-L have differences in hyperpolarization-activated, cyclic nucleotide-gated (HCN) pacemaker channels that may enable their distinct roles in behavior. Low HCN channel activity in MD-M neurons makes them relatively sensitive and responsive to inputs, while higher HCN channel activity makes MD-L neurons more specific and selective for which inputs it transforms into outputs. Knockout (KO) of the fragile X syndrome-related gene Fmr1 decreased the activity of HCN channels in MD-L but not MD-M neurons, making MD-L neurons more like MD-M cells physiologically. We sought to understand if these Fmr1-related changes in MD function were accompanied by changes in subcellular structure. Here, we assessed the dendritic morphology of MD neurons in adult Fmr1 KO and wildtype (WT) littermate mice. In acute ex vivo brain slices, we filled MD neurons with biocytin via whole cell recording pipettes. We used Neurolucida to trace the dendrites, performed quantitative analysis of dendritic architecture, compared MD-M and MD-L neurons in wildtype mice, and compared WT to KO mice for each subnucleus. In WT mice, MD-M neurons showed more distal dendritic branching compared with MD-L neurons. In Fmr1 KO mice, MD-M neurons lost their distal dendritic branching and exhibited significant reductions in multiple measures of dendritic complexity compared to WT controls (total dendritic length, number of nodes, and number of terminals), while MD-L neurons showed no morphological differences between WT and KO animals. Notably, these morphological changes show a double dissociation with our previous physiological findings: MD-M neurons exhibit structural but not functional changes, while MD-L neurons show functional but not structural changes in Fmr1 KO mice. Together, these findings suggest that FMRP regulates structure and function through independent, circuit-specific mechanisms in thalamocortical neurons, with implications for understanding the diverse cellular and network consequences of Fmr1 loss in fragile X syndrome.

Authors

Institutions

Publication Details

Journal
Brain Structure and Function
Published
2026-10-14
DOI
https://doi.org/10.1007/s00429-026-03179-5
Primary Topic
Genetics and Neurodevelopmental Disorders
Type
article
Field-Weighted Citation Impact
0.00

Funders

Controls
|||
ALL TIME
JAN
FEB
MAR
APR
MAY
JUN
JUL
AUG
SEP
OCT
article

Loss of Fmr1 causes subnucleus-specific decreases in dendritic complexity in mediodorsal thalamus

Polina N. Lyuboslavsky, Audrey C. Brumback, Alena Kizimenko
Brain Structure and Function
Genetics and Neurodevelopmental Disorders
article

Loss of Fmr1 causes subnucleus-specific decreases in dendritic complexity in mediodorsal thalamus

Polina N. Lyuboslavsky, Audrey C. Brumback, Alena Kizimenko
article en

Abstract

In the mediodorsal thalamus (MD), neurons of the medial (MD-M) and lateral (MD-L) subnuclei project to the medial prefrontal cortex (mPFC) to control complementary aspects of behavior, with MD-M supporting more of the social/emotional domain and MD-L promoting cognitive functions. Our recent work shows that MD-M and MD-L have differences in hyperpolarization-activated, cyclic nucleotide-gated (HCN) pacemaker channels that may enable their distinct roles in behavior. Low HCN channel activity in MD-M neurons makes them relatively sensitive and responsive to inputs, while higher HCN channel activity makes MD-L neurons more specific and selective for which inputs it transforms into outputs. Knockout (KO) of the fragile X syndrome-related gene Fmr1 decreased the activity of HCN channels in MD-L but not MD-M neurons, making MD-L neurons more like MD-M cells physiologically. We sought to understand if these Fmr1-related changes in MD function were accompanied by changes in subcellular structure. Here, we assessed the dendritic morphology of MD neurons in adult Fmr1 KO and wildtype (WT) littermate mice. In acute ex vivo brain slices, we filled MD neurons with biocytin via whole cell recording pipettes. We used Neurolucida to trace the dendrites, performed quantitative analysis of dendritic architecture, compared MD-M and MD-L neurons in wildtype mice, and compared WT to KO mice for each subnucleus. In WT mice, MD-M neurons showed more distal dendritic branching compared with MD-L neurons. In Fmr1 KO mice, MD-M neurons lost their distal dendritic branching and exhibited significant reductions in multiple measures of dendritic complexity compared to WT controls (total dendritic length, number of nodes, and number of terminals), while MD-L neurons showed no morphological differences between WT and KO animals. Notably, these morphological changes show a double dissociation with our previous physiological findings: MD-M neurons exhibit structural but not functional changes, while MD-L neurons show functional but not structural changes in Fmr1 KO mice. Together, these findings suggest that FMRP regulates structure and function through independent, circuit-specific mechanisms in thalamocortical neurons, with implications for understanding the diverse cellular and network consequences of Fmr1 loss in fragile X syndrome.

Brain Structure and FunctionVol. 231(8)
The University of Texas at Austin (US)
University of Texas System, Pediatric Epilepsy Research Foundation, National Institutes of Health, Dell Medical School, University of Texas at Austin, College of Natural Sciences, University of Texas at Austin, National Institute of Mental Health, National Institute of Neurological Disorders and Stroke
Good health and well-being
Openalex Percentile: Top 15%
Genetics and Neurodevelopmental Disorders
AI Navigator

Ask Laika to Summarize, Analyze, and Connect papers live on the map.

Summarize Papers & Methodologies

Extract key findings, datasets, and comparative methods across publications.

Benchmark Rankings & Visual Analytics

Rank top research institutions, authors, funders, topics, and journals by Field-Weighted Citation Impact (FWCI) and paper volume with instant charts.

Connect Distant Disciplines

Bridge topological clusters on the map to find hidden collaborative intersections.