WDR45 (Q9Y484) variants and mutations

WDR45 (also known as Q9Y484) is a human protein-coding gene encoding a WD repeat domain phosphoinositide-interacting protein 4 protein. It participates in early autophagosome formation and cellular recycling pathways, with neurons particularly vulnerable to its dysfunction. De novo or mosaic loss-of-function variants cause beta-propeller protein-associated neurodegeneration, with childhood developmental delay followed by progressive dystonia, parkinsonism, and brain iron accumulation. This analysis covers 632 WDR45 variants and mutations. Of these, 71% have computational variant effect predictions. Disease context includes neurodegeneration with brain iron accumulation 5, Dystonia, and hereditary disease. Example WDR45 variants include M1I, M1K, and M1T.

Variant analysis overview

Variant and mutation evidence

Clinical, disease, and population context

Data sources

Evidence in this analysis draws on EBI Proteins Variation, UniProt, gnomAD v4, EuropePMC, 3D Hotspot Analysis, Interaction Network Analysis, Protein Data Bank, AlphaFold DB, gnomAD constraint, Open Targets, ClinGen, MaveDB, LitVar.

Notable WDR45 variants

Examples include M1I, M1K, M1T, M1V, T2I, Q3*, Q3H, Q4*. Listed records include available protein-change notation, database identifiers, clinical classifications, computational predictions, population evidence, experimental measurements, and disease context.