Aquilegia is a popular garden plant in the northern hemisphere as well as a native plant in the UK and continental Europe. In 2000, Semiaquilegia adoxoides was found infected by downy mildew in Korea, and since 2013 there have been several confirmed records of a Peronospora sp. affecting Aquilegia in the UK, with symptomatic plants being observed several years prior. Symptoms include yellow patches delineated by the veins on the leaves of affected plants, but systemically infected plants were also recorded, which are generally chlorotic and show curled leaf margins. A greyish down of conidiophores and conidia was observed on the lower side of infected leaves. Preliminary molecular phylogenetic analyses could not identify the causal agent at the species level, but revealed its affinities to other Peronospora species parasitic on the Ranunculales and Saxifragaceae. To our knowledge, this is the first occurrence of a downy mildew affecting a species of Aquilegia. Already, about 1 year after its confirmed first occurrence in the UK and 3 years after reported symptoms, a huge impact on infested gardens and nurseries has occurred. As oospore production has been observed and the pathogen can grow systemically, rendering seed transmission likely, this pathogen should be classified as a high risk pathogen for Aquilegia. Appropriate quarantine measures should be taken to restrict the pathogen from spreading.