Osteoarthropathy in congenital sensory neuropathy with anhidrosis (CSNA) is not only a rare complication but also difficult to treat. Two patients with CSNA, a three-year-old boy and a 24-year-old woman, were treated as follows. The boy was affected in his left knee and left heel and required reduction of recurrent bilateral hip dislocations. The woman was treated by amputation of her left leg above the knee because of ischemic necrosis, secondary to knee dislocations.