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[Preprint]. 2023 Jun 11:2023.06.11.544500. [Version 1] doi: 10.1101/2023.06.11.544500

Developmental transcriptional control of mitochondrial homeostasis is required for activity-dependent synaptic connectivity

Iryna Mohylyak, Mercedes Bengochea, Carlos Pascual-Caro, Noemi Asfogo, Sara Fonseca-Topp, Natasha Danda, Zeynep Kalender Atak, Maxime De Waegeneer, Pierre-Yves Plaçais, Thomas Preat, Stein Aerts, Olga Corti, Jaime de Juan-Sanz, Bassem A Hassan
PMCID: PMC10274921  PMID: 37333418

Abstract

During neuronal circuit formation, local control of axonal organelles ensures proper synaptic connectivity. Whether this process is genetically encoded is unclear and if so, its developmental regulatory mechanisms remain to be identified. We hypothesized that developmental transcription factors regulate critical parameters of organelle homeostasis that contribute to circuit wiring. We combined cell type-specific transcriptomics with a genetic screen to discover such factors. We identified Telomeric Zinc finger-Associated Protein (TZAP) as a temporal developmental regulator of neuronal mitochondrial homeostasis genes, including Pink1 . In Drosophila , loss of dTzap function during visual circuit development leads to loss of activity-dependent synaptic connectivity, that can be rescued by Pink1 expression. At the cellular level, loss of dTzap/TZAP leads to defects in mitochondrial morphology, attenuated calcium uptake and reduced synaptic vesicle release in fly and mammalian neurons. Our findings highlight developmental transcriptional regulation of mitochondrial homeostasis as a key factor in activity-dependent synaptic connectivity.

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