BACKGROUND: Acute necrotizing encephalopathy (ANE) is a rapidly progressive 
encephalopathy seen commonly in children triggered by various prodromal viral 
infections, most common being influenza virus and Human herpes virus-6.
OBJECTIVE: We report two rare cases of ANE preceded by Chikungunya infection.
CASES: A 13-year old girl presented with a three-day history of headache, fever, 
seizures, and altered sensorium. Another 42-year old man presented with two days 
history of fever and altered sensorium. Both were suspected to have viral 
encephalitis. Evaluation revealed serum positivity for Chikungunya virus. In 
both cases, diagnosis was clinched by characteristic bilateral symmetrical 
thalamic lesions with central necrosis and hemorrhage along with lesions in 
cerebral white matter, brainstem, and cerebellum.
CONCLUSIONS: ANE is reported to have high morbidity and mortality. To the best 
of our knowledge, this is the first report of ANE post-Chikungunya infection. 
Apart from being rare etiologically, the patients had excellent response to 
steroids.