Background Takayasu arteritis (TAK) is a rare chronic inflammatory vasculitis predominantly affecting the aorta and its main branches. Takayasu arteritis has been shown to increase cardiovascular risk. Supervised exercise training (SET) is a well-recognized and effective therapeutic tool improving walking performances in patients with chronic atherosclerotic disease; however, the effects of SET, and the underlying mechanisms, remain poorly documented in TAK patients. Case summary We reviewed the literature and investigated the effects of a 12-week SET programme on walking performances, physical function, and calf muscle oxygen saturation (StO2; assessed by near-infrared spectroscopy) during exercise in a 28-year-old man with TAK and symptoms of arterial lower limb claudication. The literature review evidences only two recent publications suggesting that exercise training is effective and well-tolerated in patients with arteritis. The treadmill pain-free (þ22%) and maximal (þ273%) walking distance, 6-min walking distance (þ66%), and physical function of lower extremities (þ20%) following SET were significantly improved in our patient. Moreover, we observed a greater muscle oxygen desaturation (increased oxygen extraction) during exercise. Discussion Following SET, the increased oxygen extraction may be related to improved microvascular milieu leading to a better match between muscle oxygen supply and demand during exercise. These new results may contribute to mechanistic insights in peripheral adaptations following exercise training in TAK patients and may help to explain, at least partly, the increased walking performances. Although more studies are needed to better explore the impact of exercise training, these results suggest that exercise should be recommended in TAK patients.