Objective:

This case will highlight Cheiro-Oral syndrome (COS) to increase awareness amongst clinicians and avoid delays in diagnosis and treatment.   

Background:

COS is a rare stroke syndrome, typically involving the thalamus. The ventral posterior-medial and ventral posterior-lateral nuclei are implicated in its pathogenesis. The four known types of COS have some variation of unilateral or bilateral involvement in the perioral region and fingers or hands. We describe a case of Type I COS, where a thalamic infarct leads to numbness and tingling of the contralateral perioral region and hand.  This atypical presentation can go unrecognized, delaying appropriate care. 

Design/Methods:

NA

Results:

A 63-year-old man with hypertension, hyperlipidemia, and diabetes presented for evaluation of persistent numbness and tingling involving the left perioral region and left hand/fingers. He was normotensive on arrival. He reported taking Aspirin 81 mg daily and Atorvastatin 10 mg daily at home. NIHSS 1 (sensory).  Initial imaging did not reveal any acute intracranial abnormality, large vessel occlusion, or significant stenosis. Tenecteplase was deferred due to non-disabling symptoms. He received a 300 mg Clopidogrel load and was admitted for further management. MRI brain was ordered for further evaluation and revealed an acute right posterior thalamic infarct. Based on characteristics of the area of ischemia and uncontrolled vascular risk factors, stroke was attributed to small vessel disease. Atorvastatin was increased, and he was advised on the importance of glycemic control given significantly elevated Hemoglobin A1c. He was discharged on dual antiplatelet therapy for 21 days followed by Clopidogrel monotherapy indefinitely and was advised to continue strict vascular risk factor control.   

Conclusions:

With atypical presentations, it is important to consider stroke on the differential. Prompt recognition is essential to ensure timely initiation of appropriate therapies and conservative interventions to improve future outcomes for patients.