Background and Purpose— Providing ongoing care for rare neurological conditions is challenging. Telemedicine can reduce patient travel. We set up and evaluated a telemedicine service for patients with a genetic form of stroke and dementia cerebral autosomal dominant arteriopathy with subcortical infarcts and leukoencephalopathy. Methods— One hundred fourteen patients with mutation-positive cerebral autosomal dominant arteriopathy with subcortical infarcts and leukoencephalopathy (64 telemedicine and 50 face-to-face) were recruited. Patient and clinician satisfaction questionnaires rated the service to create a mean satisfaction score for both face-to-face and telemedicine follow-up appointments. Results— There was no difference in mean (SD) patient or clinician satisfaction scores between telemedicine and face-to-face appointments (patient: 4.57 [0.56] and 4.69 [0.42]; P =0.99; clinician: 4.55 [0.49] and 4.60 [0.43]; P =0.44). Conclusions— Telemedicine follow-up was suited to patients with stroke and dementia and offered satisfaction levels similar to that for face-to-face consultations.