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Dengue fever is a common tropical illness with varying severity often causing thrombocytopenia and haemorrhagic manifestations. We describe 2 cases with central nervous system manifestations in dengue fever. First is the case of a 20-year-old female who presented with high grade fever, headache, altered sensorium, and seizures. Clinical findings were suggestive of meningoencephalitis. Initial blood investigations of the patient showed thrombocytopenia. The magnetic resonance imaging of the brain showed bilateral thalamic, internal capsule, and occipital lobe hyper-intensities. Based on the preliminary examination and investigations, a diagnosis of acute febrile illness with meningoencephalitis was made. Further investigations showed Dengue NS1 antigen positivity in serum and a raise in anti-dengue IgM antibodies. Following symptomatic treatment and antiepileptic drug usage, the patient made a complete recovery. In another case, a 32-year-old gentleman with a short duration of fever and altered sensorium was diagnosed with dengue fever based on NS1 antigen positivity and IgM antibody positivity. He had poor GCS and MRI brain revealed bilateral thalamic and cerebellar hyper-intensities suggestive of encephalitis. He succumbed to the illness. These 2 cases highlight the importance of knowing the atypical manifestation of dengue fever.