Celiac disease is an immune disorder occurring in response to ingestion of gluten in genetically predisposed individuals. It is a complex multiorgan disease with possible neurological involvement. Thrombotic events can occur but rarely as presenting symptom. We describe the case of a young man admitted to the Stroke Unit for worsening headache, nausea, vomiting, unsteadiness, dysarthria, and dysmetria. Brain CT scan showed an ischemic hypodense lesion of the right cerebellar hemisphere with compression of the adjacent IV ventricle. Studies to determine the etiology of stroke showed positive serology for celiac disease. On follow up he has completely recovered clinically. He is on gluten-free diet and the serology is now negative. Our case demonstrated that in young stroke, even without gastrointestinal symptoms, celiac disease should be considered a possible differential diagnosis and the appropriate serology test performed.