This multimodal imaging study found VS to be associated with regional structural and functional alterations in the early and higher visual cortex, as well as with hyperconnectivity to the temporal cortex. The involved brain regions are related to visual processing, memory, spatial attention, and cognitive control. Both functional and structural changes arise in VS patients, be it as an epiphenomenon or as a direct contributor to the pathomechanism of VS. The tight link of GMV in both lingual gyri to symptom duration underlines the critical role of the lingual gyrus in disease manifestation. These in vivo neuroimaging biomarkers may hold potential as objective measures of this intriguing condition, which cannot yet be objectively quantified.