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Kidney Week

Abstract: TH-PO0568

Disruption of DYRK1A in Nephron Progenitors Drives Kidney Developmental Defects

Session Information

Category: Development, Stem Cells, and Regenerative Medicine

  • 600 Development, Stem Cells, and Regenerative Medicine

Authors

  • Romero Mora, Adrian, The University of Texas Health Science Center at Houston, Houston, Texas, United States
  • Corkins, Mark E., The University of Texas Health Science Center at Houston, Houston, Texas, United States
  • Miller, Rachel Katherine, The University of Texas Health Science Center at Houston, Houston, Texas, United States

Group or Team Name

  • Miller Lab.
Background

DYRK1A, a protein kinase whose disruption underlies DYRK1A-related intellectual disability syndrome, has been implicated in congenital anomalies of the kidney and urinary tract (CAKUT) in prior studies from our group. Over 50% of individuals with DYRK1A variants exhibit renal or genitourinary defects, although the underlying mechanisms remain poorly understood. We hypothesize that DYRK1A regulates nephron progenitor survival by modulating cell cycle and apoptosis pathways, with progenitor heterogeneity leading to differential responses upon disruption.

Methods

We used Xenopus laevis as a model for kidney development due to its conserved gene expression profile and nephron cell types comparable to humans. Dyrk1a expression was characterized through immunostaining and Western blot analysis. Functional studies included morpholino-mediated knockdown and pharmacological inhibition of Dyrk1a. Additionally, single-cell RNA sequencing (scRNA-seq) was performed to investigate transcriptomic changes in nephron progenitor populations following Dyrk1a inhibition.

Results

Preliminary data indicate that Dyrk1a dynamically localizes within nephron progenitors. Disruption of Dyrk1a leads to increased apoptosis in a subset of kidney progenitor cells without change in proliferation. Rescue experiments successfully restored the phenotype through overexpression of dyrk1a, while Dyrk1a inhibition revealed a distinct population of cells absent under normal conditions, as identified through comparative scRNA-seq analysis. We will use Hybridization Chain Reaction (HCR) to further characterize this population and map the spatial distribution of specific mRNA transcripts within the affected cells.

Conclusion

The preliminary results demonstrate that DYRK1A plays a crucial role in nephron progenitor survival and kidney development. This research will enhance our understanding of DYRK1A’s role in kidney development and its impact on DYRK1A-related intellectual disability syndrome, with potential relevance to Down syndrome and other related conditions.

Funding

  • NIDDK Support

Digital Object Identifier (DOI)