Carpal tunnel syndrome (CTS) is the most frequent entrapment mono-neuropathy, a pressure-induced neuropathy of the median nerve at the wrist. Two patients with dyskinetic cerebral palsy presented tingling sense of bilateral hands and marked flattening of both thenar eminences. In two patients, the involuntary dystonic muscle contractions kept the wrist position in hyperextension or more frequently, in hyperflexion with ulnar deviation and finger in flexion strongly. We performed careful history taking and physical examination, and then diagnosed bilateral carpal tunnel syndrome in two patients through medical workup including electromyography and ultrasonography. In this report, we present these cases and discuss their physiopathology.