Development of proof-of-concept Pitt-Hopkins Syndrome therapy by upregulation of TCF4 transcriptional activity

Awardee: Tonis Timmusk

Institution: Tallinn University of Technology

Grant Amount: $71,650.00

Funding Period: February 1, 2023 - January 31, 2024


Summary:

Pitt Hopkins syndrome (PTHS) is a genetic developmental disorder that severely affects cognitive, motor and social development. PTHS has been diagnosed in less than 1000 people in the world. It is caused by mutations in one of the two alleles of a gene called TCF4, which encodes a protein named Transcription Factor 4. Most of the mutations found in TCF4 gene in PTHS patients are of de novo origin meaning that the mutation is not present in the parents. TCF4, as other transcription factors, is a protein that regulates the expression of genes. In the nervous system TCF4 plays an important role in proliferation, differentiation and migration of neurons, as well as brain plasticity. Upregulation of the transcriptional activity of the functional TCF4 protein could improve the symptoms of PTHS. The goal of the current project is to develop a method to upregulate TCF4 activity levels by targeting TCF4 co-regulators.

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Deciphering the neurobiological pathways involved in heterogenous SETBP1 haploinsufficiency disorder using human brain organoids and transcriptomics

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Fingerprinting a multiomics biomarker profile in patients with STXBP1-RD