Article

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 mu g/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 mu g of baclofen and 25 mu g 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. (c) 2005 by American Congress of Rehabilitation Medicine and the American Academy of Physical Medicine and Rehabilitation

Langue : ANGLAIS