To describe a rare presentation of aphemia, bucco-lingual apraxia following non-dominant sub-insular stroke.

Aphemia or apraxia of speech is reported in dominant frontal lobe strokes and in Foix-Chavany-Marie (FCM) syndrome with bilateral opercular involvement. Aphemia is not reported in non-dominant sub-insular strokes.

Case Report. 52-year-old White Female with +100 score on Edinburgh handedness inventory, past medical history of hypertension, uncontrolled diabetes (A1c 11), hyperlipidemia presented to us with acute onset mutism, subtle bilateral volitional and emotional facial paresis, tongue weakness, dysphagia, bucco-lingual apraxia with preserved automatic movements like yawning. She had intact comprehension and could express by writing and drawing but was unable to phonate, mouth words, count or sing. MRI Brain showed area of diffusion restriction involving right external capsule and subacute stroke in left external capsule. Further history revealed a previous stroke two months back involving left corona radiata extending to left external capsule from which she recovered with no residual deficits.

Aphemia is difficult to attribute to a non-dominant sub-insular stroke. However, in this patient with an old left sub-insular stroke, the contralateral new stroke unveiled symptoms by loss of compensation. From this case and previous reports of FCM syndrome, it is evident that bilateral sub-insular lesions involving operculum to brainstem projections may result in aphemia and bucco-lingual apraxia despite sparing of the opercular cortex responsible for face and mouth control in the motor homunculus. The differential diagnosis of aphemia should include FCM syndrome, its sub-cortical variant termed opercular-sub-opercular syndrome as well as dominant frontal lobe lesions.