TY - JOUR AU - Gavoci, Antoneta AU - Zhiti, Anxhela AU - Rusková, Michaela AU - Magiera, Maria M AU - Wang, Mengzhe AU - Ziegler, Karin A AU - Hausrat, Torben J AU - Ugwuja, Anselm I AU - Chakraborty, Shreyangi AU - Engelhardt, Stefan AU - Kneussel, Matthias AU - Balastik, Martin AU - Janke, Carsten AU - Misgeld, Thomas AU - Brill, Monika S TI - Polyglutamylation of microtubules drives neuronal remodeling. JO - Nature Communications VL - 16 IS - 1 SN - 2041-1723 CY - [London] PB - Springer Nature M1 - DZNE-2025-00735 SP - 5384 PY - 2025 AB - Developmental remodeling shapes neural circuits via activity-dependent pruning of synapses and axons. Regulation of the cytoskeleton is critical for this process, as microtubule loss via enzymatic severing is an early step of pruning across many circuits and species. However, how microtubule-severing enzymes, such as spastin, are activated in specific neuronal compartments remains unknown. Here, we reveal that polyglutamylation, a post-translational tubulin modification enriched in neurons, plays an instructive role in developmental remodeling by tagging microtubules for severing. Motor neuron-specific gene deletion of enzymes that add or remove tubulin polyglutamylation-TTLL glutamylases vs. CCP deglutamylases-accelerates or delays neuromuscular synapse remodeling in a neurotransmission-dependent manner. This mechanism is not specific to peripheral synapses but also operates in central circuits, e.g., the hippocampus. Thus, tubulin polyglutamylation acts as a cytoskeletal rheostat of remodeling that shapes neuronal morphology and connectivity. KW - Microtubules: metabolism KW - Animals KW - Tubulin: metabolism KW - Peptide Synthases: metabolism KW - Peptide Synthases: genetics KW - Motor Neurons: metabolism KW - Hippocampus: metabolism KW - Hippocampus: cytology KW - Neuronal Plasticity: physiology KW - Synapses: metabolism KW - Synaptic Transmission KW - Neurons: metabolism KW - Polyglutamic Acid: metabolism KW - Neuromuscular Junction: metabolism KW - Protein Processing, Post-Translational KW - Mice KW - Spastin: metabolism KW - Tubulin (NLM Chemicals) KW - Peptide Synthases (NLM Chemicals) KW - tubulin polyglutamylase (NLM Chemicals) KW - Polyglutamic Acid (NLM Chemicals) KW - Spastin (NLM Chemicals) LB - PUB:(DE-HGF)16 C6 - pmid:40562742 C2 - pmc:PMC12198417 DO - DOI:10.1038/s41467-025-60855-6 UR - https://pub.dzne.de/record/279358 ER -