Chikungunya is an alphavirus transmitted primarily by mosquito vectors Aedes aegypti and Aedes albopictus. Chikungunya
disease is epidemic in many parts of the world. The clinical spectrum of this disease range from fever, rash,
arthralgia to neurologic complications like encephalitis and rarely death. During the 2016 epidemic of Chikungunya in
Delhi, we came across an extremely rare neurological disorder of Opsoclonus-Myoclonus syndrome (OMS) in association
with Chikungunya infection. The association of OMS with Chikungunya infection has not been reported previously in the
literature. We report a case of a middle-aged gentleman who presented with areflexic quadriparesis and encephalopathy.
Subsequently, he developed a continuous saccadic movement of both eyes with intermittent jerking of limbs. He was
managed with supportive therapy which included mechanical ventilatory support, plasmapheresis and antiepileptic
medications with which he improved and was discharged to home on tracheostomy tube in-situ.
Key words: Chikungunya, Encephalitis, Opsoclonus-Myoclonus Syndrome
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