Purpose: Crimean-Congo hemorrhagic fever (CCHF) is a viral zoonosis that may be associated with high mortality rate. Several inflammatory mediators (interleukins, IFN-γ, TNF-α etc.) released during disease have been implied in the pathogenesis, which also cause ischemia.Case report: A 60-years old man working in livestock production presented to emergency department with vomiting, fever and impaired balance and gait. In laboratory evaluation, it was found that there was elevation in hepatic enzymes, decreased platelet and leukocyte counts. The patient underwent magnetic resonance (MR) imaging due to gait disorder, revealing restricted diffusion at pons compatible with acute infarct. The patient was admitted with the diagnosis of CCHF and ischemic stroke secondary to infection. During follow-up, no hemorrhage was observed and the patient was discharged as he achieved clinical recovery. Conclusion:In this case report, we discussed ischemic stroke developed secondary to infection in a patient with CCHF and its management in the light of literature.