TPGS1 (tubulin polyglutamylase complex subunit 1) is a subunit of the tubulin polyglutamylase complex that mediates polyglutamylation of tubulin in cilia and flagella, essential for their biogenesis and motility 1. The protein functions in microtubule organization and sperm axoneme assembly, with roles in microtubule binding and centrosome function. TPGS1 appears involved in glucose transport and cellular movement through its effects on microtubule polymerization 1. Beyond its canonical role in cilia biology, TPGS1 has emerged as a biomarker in immune-related pathologies. Dysregulation of TPGS1 was identified as NT1-specific in CD4 T-cells, where it participates in immune synapse formation and TCR signaling 2. Additionally, TPGS1 was identified as part of the RISK4LEP signature—a prospective 4-gene blood transcriptomic biomarker predicting leprosy development 4-61 months before clinical diagnosis (AUC=86.4%) 3. Cord blood DNA methylation at TPGS1 (CpG18582997) associates with body mass index trajectories from birth to early childhood, suggesting involvement in early metabolic programming 4. Rare variants in TPGS1 were identified in polymicrogyria, implicating it in Wnt signaling regulation relevant to cortical development 5. These findings suggest TPGS1 functions extend beyond microtubule biology to immune regulation, metabolic homeostasis, and neuronal development.