Heterotopic ossification (HO) is an ectopic formation of the lamellar bone in the soft tissues. Some authors have previously reported HO or calcific tendinitis of the peroneus longus tendon at the level of the cuboid bone, while the HO of the peroneus longus tendon in the retromalleolar portion has not been reported. The purpose of this report is to describe clinical, radiological, and histological features of this rare ossification and its treatment. To the best of our knowledge, this is the first report presenting a case of HO of the peroneus longus tendon, which developed in the retromalleolar portion.