DMWD is a WD repeat-containing protein that functions as a regulatory cofactor in the deubiquitinating USP12/DMWD/WDR48 complex, promoting USP12 enzymatic activity and suppressing ubiquitin-dependent protein degradation 1. The protein is ubiquitously expressed across adult tissues with prominent abundance in testes and brain, showing developmental regulation with increased protein levels during postnatal brain development 2. Within neural cells, DMWD localizes to the cytoplasm, nucleus, and dendritic synapses, concentrating in synapse-dense brain regions 2. DMWD is located upstream of the DMPK gene in the myotonic dystrophy (DM1) locus on chromosome 19. Altered DMWD expression has been observed in DM1 patients, suggesting involvement in disease manifestation, though CTG repeat expansion shows variable effects on DMWD expression compared to neighboring genes 3. Recent functional studies identified DMWD as a neuroprotective candidate that can rescue behavioral deficits and reduce tau protein accumulation in Alzheimer disease models 4, indicating broader neurobiological significance beyond DM1. In DM1 pathophysiology, dosage reduction of DMWD alongside mutations in other locus genes (DMPK, SIX5, MBNL1) contributes to multisystem manifestations, with quadruple heterozygous mutations recapitulating congenital DM1 phenotypes 5. These findings suggest DMWD participates in both normal neural function and disease pathways.