One out of four ischemic strokes in France occurs in adults under 65 years old. About a third of them remain unexplained even after an extensive etiological assessment. A large part of these unexplained strokes could be linked to illegal drug abuse, and 10 % are estimated to be directly linked to illegal drugs in some international studies. The most frequently incriminated recreational drug remains cocaine, via several mechanisms. However, several other illegal drugs, some very commonly used such as cannabis, are suspected to have an important role in neurovascular diseases. In this article, we reviewed the epidemiological, pathophysiological and clinical studies, published in the international literature over the past 30 years. The drug-caused stroke epidemiology needs to be more precisely studied, as well as the underlying mechanisms depending on each drug. This is a public health issue that affects an economically active population, as stroke is the first cause of acquired handicap in adults.