We describe 2 patients, one with
cervical dystonia (CD) combined with
focal hand dystonia (
writer's cramp) and another with idiopathic CD, who were unresponsive to oral medications and became resistant to
botulinum toxin type A and B
injections. Both patients were successfully treated with high cervical (C1-3) continuously infused intrathecal
baclofen (ITB). Neck range of motion (ROM) was measured by using a 3-dimensional electromagnetic cervical ROM system.
Pain, disability, and severity were assessed by using the Toronto Western
Spasmodic Torticollis Rating Scale (TWSTRS). The patient with CD and
writer's cramp did well on a continuous
baclofen dose of 186.1 microg/d. Her total TWSTRS score improved significantly, her electromagnetic measurements showed an increased in total neck flexion and extension, and her handwriting improved. Unfortunately, this patient (a heavy smoker) developed
small cell carcinoma of the lung and died 9 months after her pump was placed. Total TWSTRS score and electromagnetic measurements also significantly improved after pump implant in the patient with CD. He continues to do well on a periodic bolus dose using a combination of 50 microg of
baclofen and 25 microg of
hydromorphone (
Dilaudid) every 4 hours. Our findings suggest the potential usefulness of this
therapy in other patients with
focal dystonia. To our knowledge, this is the first reported successful treatment of CD and CD combined with
writer's cramp with high cervical continuously infused ITB.