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Cheiro-Oral Syndrome (COS) is a very rare neurological syndrome associated with varied etiology. We report a 53-year-old man presented with left sided perioral and ipsilateral hand/fingers burning sensation for a one-month duration. On examination, he had hypesthesia over left perioral and distal palmar aspect of all five fingers. MRI revealed subacute infarct in the posterior limb of right internal capsule adjacent to and minimally involving thalamus. He was diagnosed as CheiroOral Syndrome as a result of ischemic stroke and managed.
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