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Ciliopathy interacts with neonatal anesthesia to cause non-apoptotic caspase-mediated motor deficits

Saric, N.; Atak, Z.; Sade, C. F.; Reddy, N.; Bell, G.; Tolete, C.; Rajtboriraks, M. T.; Hashimoto-Torii, K.; Jevtovic-Todorovic, V.; Haydar, T. F.; Ishibashi, N.

2024-11-27 neuroscience
10.1101/2024.11.27.624302 bioRxiv
Show abstract

Increasing evidence suggests that anesthesia may induce developmental neurotoxicity, yet the influence of genetic predispositions associated with congenital anomalies on this toxicity remains largely unknown. Children with congenital heart disease often exhibit mutations in cilia-related genes and ciliary dysfunction, requiring sedation for their catheter or surgical interventions during the neonatal period. Here we demonstrate that briefly exposing ciliopathic neonatal mice to ketamine causes motor skill impairments, which are associated with a baseline deficit in neocortical layer V neuron apical spine density and their altered dynamics during motor learning.. These neuromorphological changes were linked to augmented non-apoptotic neuronal caspase activation. Neonatal caspase suppression rescued the spine density and motor deficits, confirming the requirement for sublethal caspase signaling in appropriate spine formation and motor learning. Our findings suggest that ciliopathy interacts with ketamine to induce motor impairments, which is reversible through caspase inhibition. Furthermore, they underscore the potential for ketamine- induced sublethal caspase responses in shaping neurodevelopmental outcomes.

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