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An adolescent girl presented with an atypical scoliotic curve, pelvic obliquity, back pain, and lower-extremity paresthesias. A workup revealed generalized primary torsion dystonia. The condition was refractory to medical treatment and necessitated implantation of a deep brain stimulator. The scoliosis required operative correction, and the patient underwent posterior spinal arthrodesis with hook-rod instrumentation, which resulted in successful correction through 7 years of follow-up.The differential diagnosis for adolescent scoliosis should include dystonia as a potential cause, especially when a patient presents with muscular contractures, an atypical scoliotic curve, pelvic obliquity, or changing curve characteristics.