Background and Purpose- Accumulating evidence has demonstrated hemodynamic abnormalities and cerebral hypoperfusion in patients with cerebral autosomal dominant arteriopathy with subcortical infarcts and leukoencephalopathy (CADASIL). Increased venous susceptibility assessed by susceptibility weighted imaging and mapping has been shown to indicate a decrease in venous oxygen saturation. This study aimed to investigate whether altered venous oxygen saturation is related to clinical phenotypes of CADASIL patients. Methods- Using 7.0-T susceptibility weighted imaging and mapping, we compared venous susceptibility of cortical veins between 41 CADASIL patients and 43 age- and sex-matched healthy controls. The magnetic resonance imaging lesion load, mini-mental state examination score, Barthel Index, and modified Rankin Scale were examined in the patient group, and the correlations between venous susceptibility and clinical characteristics were analyzed. Results- Venous susceptibility increased with age (r=0.508, P=0.001) and was higher in CADASIL patients than in healthy controls (t=-4.673; P<0.001). We found a positive association between venous susceptibility and the age-related white matter change scores (r=0.364; P=0.019), number of lacunar infarctions (r=0.520; P<0.001), number of cerebral microbleeds (rho=0.445; P=0.004), and small-vessel disease scores (rho=0.465; P=0.002) in CADASIL patients. Moreover, increased venous susceptibility was associated with higher modified Rankin Scale scores in CADASIL patients after adjustment for age- and small-vessel disease scores (odds ratio=3.178; 95% CI, 1.101-9.179; P=0.033). Conclusions- Our findings indicate that extensive cerebral hypoperfusion may induce central nervous system impairment in CADASIL, and susceptibility weighted imaging and mapping could be used clinically to assess the condition of CADASIL patients.