Five patients with generalized dystonia who were refractory to oral medications were treated by continuous intrathecal baclofen infusion. Dystonia was related to cerebral palsy in three patients and to Hallervorden-Spatz disease in two. Responsiveness to intrathecally administered baclofen was evaluated after bolus injections in one patient and during continuous infusions via an external micropump in four. Patients who responded to trial injections were subsequently implanted with a programmable pump for continuous infusion of baclofen. Dystonia in the three patients were cerebral palsy was substantially improved by continuous intrathecal baclofen infusion in doses of 500 to 800 micrograms/d. Benefit has persisted for > 19 months of continuous infusion. Dystonia in the two patients with Hallervorden-Spatz disease was not improved, although the screening trial was limited by side effects in one patient and by meningitis in the other. We conclude that continuous intrathecal baclofen infusion is beneficial therapy for some patients with generalized dystonia and that additional investigations are indicated.
Intrathecal baclofen (ITB) infusion has been shown to be an effective treatment for spasticity secondary to both cerebral palsy and spinal cord injury. Its effect on the ambulatory status of individuals with cerebral spasticity, however, has not previously been addressed. We reviewed the effect of ITB on functional ambulation in 24 patients who were ambulatory to some extent, either with or without assistive devices. Twenty-one pumps were placed in patients with spastic cerebral palsy and 3 in patients with spasticity secondary to traumatic brain injury (13 boys and 11 girls, mean age 18 years). The mean ITB dose was 200 µg/day (range 22–550 µg/day) and the mean length of follow-up was 52 months. Ambulation was retrospectively graded on four functional levels: community, household, non-functional, and non-ambulatory. The level of ambulation improved by one functional level in 9 patients, did not change for 12 patients, and was worse in 3 patients. Gait was considered to be improved in 20 of 24 patients by the patients or their families. The overall functional improvement not directly related to ambulation was found to be improved in 20 patients, unchanged in 2 patients, and worse in 2 patients. ITB allows for improved ambulation in a certain subset of patients with lower extremity spasticity. It is not contraindicated in patients who rely upon their spasticity for support during ambulation. ITB infusion allows for baclofen dosage titration to balance between extensor tone for support and suppression of hyperactive reflexes which may impede normal locomotion.
This study was performed to compare the effects of continuous intrathecal baclofen infusion (OBI) and selective posterior rhizotomy (SPR) on upper extremity (UE) spasticity and range of motion in children with cerebral palsy. Spasticity was assessed with the Ashworth scale of muscle tone and range of motion was evaluated. Thirty-eight patients who had been treated with CIBI for at least 6 months were paired, according to pretreatment UE muscle tone and functional status, with 38 patients who had undergone SPR. The CIBI dosage had been titrated to reduce lower extremity spasticity and improve lower extremity function, rather than to improve UE tone. The pretreatment muscle tone in the two groups was virtually identical. The UE tone of children treated with CIBI decreased from 2.07 prior to treatment to 1.66 after 1 year (p < 0.01). The tone of children treated with SPR decreased from 2.03 to 1.70 after 1 year (p = 0.005). In that group, the likelihood of a clinically significant reduction in muscle tone (one point or greater) was greater in children with a higher pretreatment UE muscle tone. There was no correlation between the percentage of posterior lumbar roots divided in SPR and the subsequent reduction in UE tone. There were no significant changes in the range of motion in any UE joint, at either 6 or 12 months, after either CIBI or SPR. We conclude that both CIBI and SPR significantly reduce UE spasticity, in addition to the previously documented reduction in lower extremity spasticity.
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