Dissertations and Theses

Date of Award

2023

Document Type

Thesis

Department

Biology

First Advisor

Hysell V. Oviedo

Second Advisor

Mark Emerson

Third Advisor

Pinar Ayata

Keywords

mismatch repair, parvalbumin, connexin36, neural circuitry, primary auditory cortex

Abstract

DNA repair mechanisms are crucial for both cellular development and function. One highly conserved DNA repair factor is Mut-S Homolog 2 (Msh2), which corrects base-base mismatches and insertion/deletion loops. In humans, defects in this repair pathway are linked to diseases that have severe neurological pathologies. These include Lynch syndrome, Huntington’s disease and demyelination of the corpus callosum. The fundamental role of Msh2 in brain function is unknown. Using an Msh2-/- mouse model we began an exploration of its impact on the processing of sensory information, a crucial function to an animal’s survival. The goal of this study was to investigate potential deficits in thalamo-cortical function. Using sound stimulation and immunohistochemical detection of active neurons with the immediate early gene cfos, we observed a significant decrease in cortical and thalamic cfos expression in Msh2-/- mice, indicating a reduction in neural activity compared to WT littermates. To examine possible mechanisms underlying this hypoactivity, we focused on the thalamic reticular nucleus (TRN): a critical regulator of corticothalamic and thalamocortical activity. We observed an increase in parvalbumin (PV+) expression, density, and cell area in the TRN, as well as high aggregation of the gap junction protein connexin-36 (Cx36). Cx36 is required for electrical coupling between TRN neurons, regulating the degree of synchrony in firing patterns. This indicates that Cx36- expressing PV+ cells in the TRN undergo increased gap junction coupling, thus leading to dysfunction in the electrical properties of the TRN, and thalamo-cortical sensory transmission. These findings suggest that Msh2, and therefore mismatch-repair, plays a role in the regulation or development of inhibitory interneurons in the TRN, leading to a downstream decrease of sound evoked neural activity.

Share

COinS
 
 

To view the content in your browser, please download Adobe Reader or, alternately,
you may Download the file to your hard drive.

NOTE: The latest versions of Adobe Reader do not support viewing PDF files within Firefox on Mac OS and if you are using a modern (Intel) Mac, there is no official plugin for viewing PDF files within the browser window.