DYNC1I2 (dynein cytoplasmic 1 intermediate chain 2) encodes a non-catalytic accessory component of the cytoplasmic dynein 1 complex that mediates cargo binding and dynein motor function 1. As an intermediate chain, DYNC1I2 links dynein to dynactin and adapter proteins, facilitating retrograde intracellular transport of vesicles and organelles along microtubules, including Golgi apparatus, late endosomes, and lysosomes 1. DYNC1I2 specifically interacts with TMEM39A to regulate lysosome distribution and positioning in cells 2. Bi-allelic DYNC1I2 variants cause autosomal-recessive neurodevelopmental disorder with microcephaly and structural brain anomalies 13. Pathogenic variants impair neurogenesis through disrupted cargo transport essential for neuronal development 1. Functional studies in zebrafish demonstrate that dync1i2 dysfunction causes abnormal spindle morphology, increased apoptosis during prolonged mitosis, and altered craniofacial patterning with reduced head size 1. At the molecular level, DYNC1I2 is regulated post-transcriptionally; the lncRNA HHIP-AS1 protects DYNC1I2 mRNA from miR-425-5p-mediated degradation, enabling pro-mitotic effects in SHH-driven tumors 4. Alternative splicing generates tissue-specific DYNC1I2 isoforms with distinct roles in neuronal dynein complexes 5. These findings establish DYNC1I2 as critical for both normal neurodevelopment and microtubule-dependent cellular processes.