Dengue infection is common in 129 countries including Pakistan, with an estimated 390 million dengue virus infections per year. Asia accounts for 70% of the actual burden. Clinical signs and symptoms of dengue include a high fever (40°C/104 °F) accompanied by two of the following symptoms during the febrile phase: severe headache, pain behind the eyes, muscle and joint pains, nausea, vomiting, swollen glands and rash. Severe dengue is a potentially fatal complication, due to plasma leaking, fluid accumulation, respiratory distress, severe bleeding or organ impairment. Dengue virus does not usually cause neurological manifestations, unlike other arboviral infections. Encephalopathy is the most common neurological complication. Encephalitis is rare manifestation. We report a rare case of dengue encephalitis from Pakistan, with extensive brain lesions involving the thalamus, cerebellar vermis and frontoparietal periventricular regions. Therefore, we suggest dengue encephalitis should be considered in the differential diagnosis of fever with altered sensorium, especially in Asian countries such as Pakistan where dengue is rampant as dengue encephalitis can be fatal at times.