The use of standard anterolateral and anteromedial portals in ankle arthroscopy results in reduced risk of vascular complications. Anatomical variations of the arterial network of the foot and ankle might render the vessels more susceptible to injury during procedures involving the anterior ankle joint. The literature, to our knowledge, reports only one case of a pseudoaneurysm involving the peroneal artery after ankle arthroscopy. Here, we report the unusual case of a 48-year-old man in general good health with the absence of the anterior tibial artery and posterior tibial artery. The patient presented with a pseudoaneurysm of the perforating peroneal artery following ankle arthroscopy for traumatic osteoarthritis associated with nonunion of the medial malleolus. The perforating peroneal artery injury was repaired by performing end-to-end anastomosis. The perforating peroneal artery is at higher risk for iatrogenic injury during ankle arthroscopy in the presence of abnormal arterial variations of the foot and ankle, particularly the absence of the anterior tibial artery and posterior tibial artery. Before ankle arthroscopy, surgeons should therefore carefully observe the course of the perforating peroneal artery on enhanced 3-dimensional computed tomography, especially in patients with a history of trauma to the ankle joint.