Headaches in Moyamoya angiopathy are common but poorly understood. We aimed to investigate if headaches in Moyamoya angiopathy improve after revascularization surgery and whether this is associated with improvement in cerebrovascular reactivity on MRI (CVR-MRI). We included consecutive adult patients with Moyamoya angiopathy who had chart data on headaches, CVR-MRI, and underwent extracranial-intracranial bypass surgery between January 2010 and September 2022 at a tertiary neurovascular referral center. Clinical and CVR-MR imaging data of all patients were collected through systematic chart review, complemented by standard-of-care headache questionnaires from patients who were operated between 2018 and 2022. We evaluated headache features and explored the association between headaches and CVR before and after revascularization surgery. Fifty-nine patients were included (mean age 47 ± 14 years, 43 females (73%)); among them, 41/59 (69%) reported headaches pre-surgery. Headache improved in 28/41 (68%) patients after revascularization surgery with a reduction in pain severity (median VAS-score from 5/10 to 2.5/10; p = 0.002), analgesic use (from 84% to 40%; p = 0.007), and sick leave (from 60% to 16%; p < 0.001). Improvement in headaches was associated with improvement in CVR (OR 5.3; 95% CI: 1.2-23.5) and sick leave reduction (OR 1.4; 95% CI: 1.6-121.4). Headaches in Moyamoya angiopathy are common and disabling. They may improve in most patients after revascularization surgery and seem to be associated with improvement in CVR, supporting the hypothesis of a potential vascular origin of the headaches.