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Background and Purpose: To characterize clinically acute external (EcC) and extreme capsular (ExC) strokes in 9 patients with first-ever acute stroke. Methods: Nine patients with a lesion limited to the EcC-ExC region proven by diffusion-weighted imaging included in our registry, corresponding to 0.3% of 3,600 patients with ischemic stroke, were studied. Results: We found three main groups of clinical manifestations: (1) motor deficits in all patients (4 with faciobrachial, 2 with faciobrachiocrural, and 1 with brachiocrural, crural and facial paresis each); (2) hypesthesia in 3 patients with partial distribution concerning light touching without involvement of other sensory modalities; (3) transient speech disturbance at stroke onset in 1 patient and dysarthria in 2 patients. Small artery disease in 4 patients and artery-to-artery embolism in 3 patients probably from atherosclerotic carotid artery were the main stroke mechanism. Conclusions: Strokes restricted to the EcC-ExC region may present with complete or partial motor syndromes and dysarthria as prominent clinical presentations, but also fou rire prodromique and transient speech arrest as prodromal signs. The main etiology of stroke were microangiopathy and artery-to-artery embolism, even though no obvious cause was found in 1 patient. The prognosis was good in almost all patients.