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Identifying the neural underpinnings of socio-emotional deficits in 22q11.2 deletion syndrome (22q11DS) may elucidate recurrent psychosis in affected individuals. In the current study, we investigate volumetric changes in 22q11DS to the fusiform gyrus (FG), a region associated with hypoactivity during fMRI, by manually tracing the FG in 42 individuals with 22q11DS and 54 healthy controls. Larger anterior FG volumes and smaller posterior volumes bilaterally are observed in 22q11DS after controlling for total brain volume. The results demonstrate structural changes to the FG in 22q11DS, providing evidence for neural vulnerability in regions related to social cognition.