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Background Patients with reflex sympathetic dystrophy (also known as the complex regional pain syndrome) may have dystonia, which is often unresponsive to treatment. Some forms of dystonia respond to the intrathecal administration of baclofen, a specific
Methods We performed a double-blind, randomized, controlled crossover trial of bolus intrathecal injections of 25, 50, and 75 µg of baclofen and placebo. Changes in the severity of dystonia were assessed by the woman and by an investigator after each injection. In the second phase of the study, six of the women received a subcutaneous pump for continuous intrathecal administration of baclofen and were followed for 0.5 to 3 years.
Results In six women, bolus injections of 50 and 75 µg of baclofen resulted in complete or partial resolution of focal dystonia of the hands but little improvement in dystonia of the legs. During continuous therapy, three women regained normal hand function, and two of these three women regained the ability to walk (one only indoors). In one woman who received continuous therapy, the pain and violent jerks disappeared and the dystonic posturing of the arm decreased. In two women the spasms or restlessness of the legs decreased, without any change in the dystonia.
Conclusions In some patients, the dystonia associated with reflex sympathetic dystrophy responds markedly to intrathecal baclofen. (N Engl J Med 2000; 343:625-30.)
-aminobutyric acid–receptor (type B) agonist that inhibits sensory input to the neurons of the spinal cord. We evaluated this treatment in seven women who had reflex sympathetic dystrophy with multifocal or generalized tonic dystonia.
Source Information
From the Departments of Neurology (B.J.H., W.-J.T.B., J.I.H., E.M.D.) and Neurosurgery (J.H.C.V.), Leiden University Medical Center, Leiden, the Netherlands.
Address reprint requests to Dr. van Hilten at the Department of Neurology, Leiden University Medical Center, P.O. Box 9600, 2300 RC Leiden, the Netherlands, or at jvhilten{at}lumc.nl.
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