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PubMed1994

Effects of selective dorsal rhizotomy for spastic diplegia on hip migration in cerebral palsy.

In spastic diplegia of cerebral palsy, migration of the femoral head beyond a lateral edge of the acetabulum is a common orthopedic deformity and requires surgical treatment. We investigated whether selective dorsal rhizotomy for spastic diplegia halts or exacerbates lateral hip migration. The Reimers migration percentage computed from preoperative and postoperative hip radiographs was used as an index of the severity of lateral hip migration in all 134 hips of 67 children examined. At the time of rhizotomy, 38 patients were between 2 and 4 years of age and 29 were between 5 and 11 years of age. The follow-up period ranged from 6 to 10 months in 20 patients and from 15 to 46 months in 47 patients. Overall, the MP remained unchanged in 75% decreased in 17% and increased in 7%; thus, 93% of all hips examined were stable radiographically. Although most patients experienced postoperative hip stability, there was a significant trend for patients with greater preoperative migration to show decreased postoperative migration. The preoperative ambulatory status of patients had no impact on hip stability after dorsal rhizotomy. By the last follow-up, only 1 patient had undergone orthopedic operations for a persistent hip deformity. The results suggest that in children with spastic diplegia, selective dorsal rhizotomy halts lateral hip migration in the great majority of cases.

Adolescent↗
PubMed1995

Cost analysis of continuous intrathecal baclofen versus selective functional posterior rhizotomy in the treatment of spastic quadriplegia associated with cerebral palsy.

The purpose of the study was to analyze the relative cost of selective functional posterior rhizotomy (SFPR) and continuous intrathecal baclofen in the treatment of children with severe spastic quadriplegia related to cerebral palsy. No attempt was made to analyze the efficacy of the two types of treatment. Nine children with spastic quadriplegia secondary to cerebral palsy in whom continuous intrathecal baclofen was attempted were matched as closely as possible with a group of 10 patients with spastic quadriplegia out of a total of 100 children who had undergone SFPR in the same time period. Clinical care flow charts were created to identify the various points of contact with members of the health care team, so that cost points could be identified and costs calculated. The cost per patient up to 1 year a after treatment CDN$ 64,163.10 for patients with implanted pumps for continuous intrathecal baclofen versus CDN$ 16,913.54 for SFPR. When adjustments were made to exclude costs and savings associated with research protocols, the average for the baclofen group decreased to approximately CDN$ 63,000, with minimal change for the SFPR group. The higher cost per patient on baclofen was related to the cost associated with screening patients who did not go on to have implantation of a continuous infusion pump, and to additional hospitalization for complications in the baclofen group. It is cautioned that this cost analysis was based on the experience at British Columbia's Children's Hospital, and the results may not be generalizable to other institutions or to other patient populations.

Adolescent↗
PubMed2000

A randomized, double-blind, placebo-controlled, dose-ranging study to compare the efficacy and safety of three doses of botulinum toxin type A (Dysport) with placebo in upper limb spasticity after stroke.

BACKGROUND AND PURPOSE: We sought to define an effective and safe dose of botulinum toxin type A (Dysport) for the treatment of upper limb muscle spasticity due to stroke. METHODS: This was a prospective, randomized, double-blind, placebo-controlled, dose-ranging study. Patients received either a placebo or 1 of 3 doses of Dysport (500, 1000, 1500 U) into 5 muscles of the affected arm. Efficacy was assessed periodically by the Modified Ashworth Scale and a battery of functional outcome measures. RESULTS: Eighty-three patients were recruited, and 82 completed the study. The 4 study groups were comparable at baseline with respect to their demographic characteristics and severity of spasticity. All doses of Dysport studied showed a significant reduction from baseline of muscle tone compared with placebo. However, the effect on functional disability was not statistically significant and was best at a dose of 1000 U. There were no statistically significant differences between the groups in the incidence of adverse events. CONCLUSIONS: The present study suggests that treatment with Dysport reduces muscle tone in patients with poststroke upper limb spasticity. Treatment was effective at doses of Dysport of 500, 1000, and 1500 U. The optimal dose for treatment of patients with residual voluntary movements in the upper limb appears to be 1000 U. Dysport is safe in the doses used in this study.

Arm↗
PubMed1998

Manual dexterity and keyboard use in spastic hemiparesis: a comparison between the impaired hand and the 'good' hand on a number of performance measures.

OBJECTIVE: To assess the functional deficits in tapping performance of subjects with spastic hemiparesis. DESIGN: In a pilot study, typing performance on a computer keyboard was examined on a number of performance measures. SETTING: Department of Research and Development at the Werkenrode Institute. SUBJECTS: Four subjects (mean age 16.4 years, standard deviation 1.8 years) with cerebral palsy and diagnosed as having spastic hemiparesis. INTERVENTIONS: Subjects had to type a sequence of one or more keys as quickly as possible within an 8 second period with the fingers of both hands separately. MAIN OUTCOME MEASURES: The average number of good and false responses were calculated. Within the false response category, four, mutually exclusive, types of errors were distinguished; wrong key, repetition, registrations under 75 ms and holding. Speed and regularity of the typing responses were also established. RESULTS: The 'good' hand outperformed the impaired hand on all performance measures except on the amount of repetition errors made. The holding error was only present for the impaired hand, and there was an increase in holding errors from the index to the little finger in this hand. In addition, the impaired hand performed the task with a slower speed and in a more irregular fashion. CONCLUSIONS: The results are discussed with reference to keyboard design. It is concluded that the standard 'QWERTY' keyboard hampers typing performance extensively, especially for subjects with left spastic hemiparesis. A learning method is presented in which the role of an external pacer (e.g. metronome) is discussed. This device can initially be used to decrease movement variability and, eventually, for increasing movement speed.

Adolescent↗
PubMed1989

Split posterior tibial tendon transfer for spastic equinovarus foot deformity.

This paper describes a simplified technique for split posterior tibial tendon transfer in the treatment of spastic equinovarus deformity of the foot. Thirteen children with spastic equinovarus foot deformities were treated at Children's Rehabilitative Services in Phoenix, Arizona, from 1983 to 1986. The technique was modified in 10 of the 13 patients by attaching the split posterior tibial tendon more proximally to the peroneus brevis, compared to a more distal attachment as described by other authors. The mean length of followup was 21 months. Eleven patients obtained a good or excellent result. Two patients were considered to have a fair result. No poor results or complications were noted in any of the patients. It was felt that the split posterior tibial tendon transfer was an effective procedure for correction of spastic equinovarus as reported by other authors. Modification of the technique significantly simplifies the operation by requiring less dissection while still producing favorable results.

Adolescent↗
PubMed1991

The use of open phenol blocks to the motor branches of the tibial nerve in adult acquired spasticity.

Spasticity of the posterior calf muscles often occurs following head injury or spinal cord injury. If left untreated, equinovarus contractures of the foot and ankle may occur in a relatively short time period. This paper retrospectively reviews nine patients with either closed head injury (six patients) or spinal cord injury (three patients) who underwent open phenol injections into the motor branches of the tibial nerve in 16 limbs. Spasticity was decreased in all patients. No significant equinus contractures had occurred at final follow-up. Five of the patients were subsequently able to ambulate. One patient had recurrence of spasticity requiring a second surgical procedure (split anterior tibial tendon transfer), and two patients had significant toe clawing at final follow-up. Open phenol injections to the motor branches of the tibial nerve safely and effectively prevent foot and ankle contractures following spinal cord injury or head injury.

Adult↗
PubMed2000

Baclofen increases the soleus stretch reflex threshold in the early swing phase during walking in spastic multiple sclerosis patients.

The effect of baclofen on walking performance was examined in nine spastic multiple sclerosis patients. In addition, nine healthy subjects were tested as controls. The modulation of the short latency soleus stretch reflex was closer to normal with baclofen compared to the recordings without baclofen, the modulation index being 74% (range: 60 - 100) with baclofen and 62% (range: 20 - 100) without baclofen, P=0.03. In healthy subjects the modulation index was 100% (range: 52 - 100). In the early swing phase the threshold of the soleus stretch reflex was significantly higher during baclofen medication being 139 deg/s (range: 63 - 302) compared with 93 deg/s (range: 37 - 187) without baclofen, P=0.004. The relation between the stretch velocity (input) and the amplitude of the stretch reflex (output) in early swing phase was unchanged being 0. 27 microVs/deg (range: 0.1 - 1.51) in patients with baclofen and 0. 24 microVs/deg (range: 0.08 - 0.79) without baclofen, P=0.25. Baclofen induced no change in input - output properties of the stretch reflex during walking compared with findings in a sitting position at matched EMG activity. There was a significant correlation between clinical spasticity score and stretch reflex threshold in the early swing phase (rho=-0.61, P=0.04) and between clinical spasticity score and the slope of the best linear fit in the early swing phase (rho=0.72, P=0.009).

Adult↗
PubMed1996

Treatment of spasticity with repetitive magnetic stimulation; a double-blind placebo-controlled study.

The effect of repetitive magnetic stimulation on spasticity was evaluated in 38 patients with multiple sclerosis in a double-blind placebo-controlled study. One group was treated with repetitive magnetic stimulation (n = 21) and the other group with sham stimulation (n = 17). Both groups were treated twice daily for 7 consecutive days. Primary end-points of the study were changes in the patients self-score, in clinical spasticity score, and in the stretch reflex threshold. The self-score of ease of daily day activities improved by 22% (P = 0.007) after treatment and by 29% (P = 0.004) after sham stimulation. The clinical spasticity score improved -3.3 +/- 4.7 arbitrary unit (AU) in treated patients and 0.7 +/- 2.5 AU in sham stimulation (P = 0.003). The stretch reflex threshold increased 4.3 +/- 7.5 deg/s in treated patients and -3.8 +/- 9.7 deg/s in sham stimulation (P = 0.001). The data presented in this study supports the idea that repetitive magnetic stimulation has an antispastic effect in multiple sclerosis. Future studies should clarify the optimal treatment regimen.

Activities of Daily Living↗
PubMed2000

Reciprocal tapping in spastic hemiparesis.

OBJECTIVE: To locate the source of slowness in typing movements in subjects with spastic hemiparesis and to examine whether enlargement of keys would facilitate typing. DESIGN: Experimental within-subjects design. SETTING: Werkenrode, a special school for physically disabled young children. SUBJECTS: Eleven subjects (mean age 17.1 years) diagnosed as having spastic hemiparesis, most as a result of cerebral palsy. INTERVENTIONS: Subjects made fast reciprocal tapping movements with one finger of both hands separately within a 10-second interval. Keys of 12 x 12 mm and 24 x 24 mm were used. MAIN OUTCOME MEASURES: Interkey response interval (IRI), dwell time (interval during which the key is pressed) and forces were measured. Also, the standard deviation and coefficient of variation of the variables were calculated to determine regularity of the movements. RESULTS: The impaired hand tapped slower and more irregularly and exerted less force. In addition, the duration of the dwell phase was lengthened for this hand. However, the percentage dwell time as a function of IRI was not different between hands. Enlargement of the keys shortened flight time (i.e. time in which the finger moves through the air from one key to the next) of the impaired hand, but not dwell time. CONCLUSIONS: The entire movement is slower for the impaired hand. Disturbances are not exclusively located at the inversion of the movement. With respect to keyboard design, enlarged keys and a locked repeat function of the keys may be beneficial for subjects suffering from spasticity.

Adolescent↗
PubMed2005

Taping versus electrical stimulation after botulinum toxin type A injection for wrist and finger spasticity. A case-control study.

OBJECTIVE: To compare results from two approaches used in conjunction with botulinum toxin type A administration in rehabilitation: the application of a taping system and the electrical stimulation of the injected muscles and splinting. DESIGN: Case-control study. SETTING: Two tertiary care rehabilitation hospitals in Italy. SUBJECTS: Sixty-five adult subjects affected by spasticity of the wrist and finger flexors. INTERVENTIONS: After injection with botulinum toxin type A, the group at hospital A (n=33) was treated with adhesive taping for six days and those at hospital B (n=32) with electrical stimulation and splinting for six days. Spastic hypertonia at the injected muscles was assessed before treatment, one week and one month post injection. MAIN MEASURES: Modified Ashworth Scale. RESULTS: In group A, the mean Modified Ashworth Scale reduction was 2.76 +/- 0.94 for wrist flexors and 2.45 +/- 0.92 for finger flexors; in group B the mean Modified Ashworth Scale reduction was 2.18 +/- 1.11 for wrist flexors and 2.1 +/- 0.98 for finger flexors. The observed difference between the two groups was statistically relevant (p < 0.05). CONCLUSIONS: Patients treated with adhesive taping and botulinum toxin type A achieved a greater reduction in spastic hypertonia as measured with Modified Ashworth Scale, with less time dedicated for the treatment.

Bandages↗
PubMed2002

A novel form of autosomal recessive pure hereditary spastic paraplegia maps to chromosome 13q14.

BACKGROUND: Hereditary spastic paraplegia (HSP) is a clinically and genetically heterogeneous disorder characterized by a progressive weakening and spasticity of the lower limbs. HSP is classified according to the presence or absence of accompanying neurologic problems and by the mode of inheritance. Currently, 17 loci have been linked to the various forms of HSP. OBJECTIVE: To determine the chromosomal location of a gene causing pure autosomal recessive spastic paraplegia. METHODS: Genotyping using fluorescently labeled microsatellite markers was performed on three affected individuals and three unaffected individuals from a family displaying pure autosomal recessive HSP (ARHSP) and sensorineural deafness. All family members were then included in the analysis to narrow the genetic interval. Candidate genes were screened for the presence of mutations by heteroduplex analysis. RESULTS: The paraplegic trait linked to a 1.8-Mb region of chromosome 13q14 flanked by the FLJ11712 gene and the microsatellite marker D13S270. The deafness did not link to this region and did not cosegregate with the paraplegic trait. CONCLUSION: The HSP that this family had represents a novel genetic form of pure ARHSP as no other form of HSP (autosomal dominant or recessive) has been linked to chromosome 13.

Abnormalities, Multiple↗
PubMed2003

Infantile ascending hereditary spastic paralysis (IAHSP): clinical features in 11 families.

OBJECTIVE: To report clinical, neuroradiologic, neurophysiologic, and genetic findings on 16 patients from 11 unrelated families with a remarkable uniform phenotype characterized by infantile ascending hereditary spastic paralysis (IAHSP). METHODS: Sixteen patients from 11 families, originating from North Africa and Europe, who presented severe spastic paralysis and ascending progression were studied. RESULTS: Spastic paraplegia started in the first 2 years of life in most patients and extended to the upper limbs by the end of the first decade. The disease progressed to tetraplegia, anarthria, dysphagia, and slow eye movements in the second decade. The clinical course showed a long survival and preservation of intellectual skills. Clinical, neuroradiologic, and neurophysiologic findings were consistent with a relatively selective early involvement of the corticospinal and corticobulbar pathways. No signs of lower motor neuron involvement were observed, whereas motor evoked potentials demonstrated predominant involvement of the upper motor neurons. MRI was normal in young patients but showed brain cortical atrophy in the oldest, predominant in the motor areas, and T2-weighted bilateral hyperintense signals in the posterior arm of the internal capsule. The ALS2 gene, recently found mutated in consanguineous Arabic families with either an ALS2 phenotype or a juvenile-onset primary lateral sclerosis, was analyzed. Alsin mutations were found in only 4 of the 10 families, whereas haplotype analysis excluded the ALS2 locus in one family. CONCLUSIONS: The syndrome of IAHSP is genetically heterogeneous, and no clinical sign can help to distinguish patients with and without Alsin mutations.

Adolescent↗
PubMed2006

A novel locus for hereditary spastic paraplegia with thin corpus callosum and epilepsy.

BACKGROUND: Hereditary spastic paraplegia (HSP) are classified clinically as pure when progressive spasticity occurs in isolation or complicated when other neurologic abnormalities are present. At least 22 genetic loci have been linked to HSP, 8 of which are autosomal recessive (ARHSP). HSP complicated with the presence of thin corpus callosum (HSP-TCC) is a common subtype of HSP. One genetic locus has been identified on chromosome 15q13-q15 (SPG11) for HSP-TCC, but some HSP-TCC families have not been linked to this locus. METHODS: The authors characterized two families clinically and radiologically and performed a genome-wide scan and linkage analysis. RESULTS: The two families had complicated ARHSP. The affected individuals in Family A had thin corpus callosum and mental retardation, whereas in Family B two of three affected individuals had epilepsy. In both families linkage analysis identified a locus on chromosome 8 between markers D8S1820 and D8S532 with the highest combined lod score of 7.077 at marker D8S505. This 9 cM interval located on 8p12-p11.21 represents a new locus for ARHSP-TCC. Neuregulin and KIF13B genes, located within this interval, are interesting functional candidate genes for this HSP form. CONCLUSION: Two consanguineous families with complicated autosomal recessive hereditary spastic paraplegia were clinically characterized and genetically mapped to a new locus on 8p12-p11.21.

Asian People↗
PubMed2006

Effect of intrathecal baclofen on sleep and respiratory function in patients with spasticity.

OBJECTIVE: To prospectively evaluate the effect of pump-infused intrathecal baclofen infusion (ITB) in therapeutic doses on sleep quality and on daytime and nighttime respiratory function in patients with severe spasticity. METHODS: We prospectively evaluated 20 consecutive patients (mean +/- SD age 45 +/- 13 years). We assessed spasticity and obtained polysomnography, pulmonary function tests, and resting energy expenditure measurements 1 week before and at least 8 days after pump implantation. Patients stopped oral baclofen upon pump implantation but continued other medications unchanged. We report descriptive statistics as means +/- SD. RESULTS: Most of the patients had multiple sclerosis (n = 9) or spinal cord injury (n = 8); there was one case each of cerebral palsy, hereditary spastic paraplegia, and Friedreich ataxia. Before and after ITB initiation, mean Ashworth scores were 2.75 +/- 0.85 and 1.15 +/- 0.36, and mean spasm scores were 3.75 +/- 0.55 and 1.00 +/- 0.56. ITB improved total sleep time (p = 0.05) and sleep efficiency (p = 0.01) and reduced periodic leg movements (p = 0.02). ITB did not modify sleep-related respiratory events, lung function tests, CO2 rebreathing response, or resting energy expenditure. CONCLUSION: Compared with oral baclofen, intrathecal baclofen infusion did not affect respiratory function and improved sleep continuity. Intrathecal baclofen infusion in therapeutic doses may act at the spinal level rather than at the supraspinal level.

Adult↗
PubMed1981

Familial spastic paraplegia, peroneal neuropathy, and crural hypopigmentation: a new neurocutaneous syndrome.

A neurocutaneous syndrome is described in which spastic paraplegia, peroneal neuropathy, and crural hypopigmentation are inherited in a dominant pattern. Spastic paraplegia becomes clinically apparent during adolescence or in childhood and progresses slowly throughout the adult years with a variability in severity of expression. Peroneal neuropathy is documented clinically and by slowing on nerve conduction studies. Pattern-reversal visual evoked potentials also yield a delayed response, but brainstem auditory evoked potentials and somatosensory evoked potentials in upper and lower extremities are normal. Crural hypopigmentation is present in each neurologically affected family member. Although the pattern may vary, a pretibial distribution predominates. The lower-extremity abnormalities of the central and peripheral nervous system and neural crest cell derivatives may share a common relationship, but the abnormal visual evoked potentials suggest a more generalized involvement. This family may represent a transition form between familial spastic paraplegia and the spinocerebellar degenerations.

Adult↗
PubMed1987

Sural nerve stimulation and motor control of tibialis anterior muscle in spastic paresis.

In patients with spastic paraparesis, increased extensor tonus can be decreased by stimulation of flexor reflex afferents. This should improve dorsiflexion of the foot and reduce the sense of effort. We therefore examined ability to maintain stable firing of a single motor unit (SMU) in tibialis anterior muscle and force of dorsiflexion in 17 normal subjects and 9 with spastic paresis, during several minutes of tonic nonpainful stimulation (20 Hz, 0.1 msec) of the sural nerve at the ankle (SNS). Subjects were asked to maintain stable SMU firing first with, then without auditory feedback of the motor unit potential. SNS was then applied for several minutes. In normal subjects, the force of dorsiflexion increased 33 +/- 26% with SNS and 2.5% +/- 5% without SNS (p less than 0.0005). Most subjects noted increased resistance to dorsiflexion during SNS that resulted in greater innervation of tibialis anterior muscle. In six abnormal subjects, the force of dorsiflexion increased 30 +/- 30% with SNS, but no increase was recorded without SNS. In normal subjects and those with spastic paresis of the legs, SNS increased innervation of tibialis anterior muscle and awareness of greater effort required to maintain constant innervation. The altered proprioception may depend on facilitation of motor neurons.

Electric Stimulation↗
PubMed1996

Refined genetic mapping and proteolipid protein mutation analysis in X-linked pure hereditary spastic paraplegia.

X-linked hereditary spastic paraplegias (HSP) present with two distinct phenotypes, pure and complicated. The pure form is characterized by spasticity and gait difficulties but lacks the additional features (nystagmus, dysarthria, mental retardation) present in the complicated form. The complicated form is heterogeneous, caused by mutations of the L1CAM gene at Xq28 (SPG1) or the PLP gene at Xq22 (SPG2) that is allelic to Pelizaeus-Merzbacher disease (PMD). Since in one kindred (K313) the pure form of HSP was also mapped to Xq22, this raises the issue as to whether a pure form of HSP exists that is allelic to X-linked complicated HSP (SPG2) and PMD. To answer this question, we carried out linkage analysis in a new pedigree with pure HSP (K101) and refined linkage in pedigree K313. The PLP gene was also screened for mutation by direct sequencing and reverse-transcriptase polymerase chain reaction (RT-PCR). In both families, the disease locus mapped to Xq22 with Lod scores at zero recombination of 5.3 for COL4A5 2B6 in K313 and 2.4 for DXS101 in K101. A T to C transition in exon 5 of the PLP gene was identified from affected individuals of K313. This transition causes a Ser to Pro mutation in the major extracellular loop of PLP/DM20. This finding demonstrates that a form of X-linked pure spastic paraplegia, X-linked complicated HSP (SPG2) and PMD are allelic disorders. There was no evidence of mutations in either coding sequences or the intron/exon junctions of PLP in pedigree K101, suggesting that the disease-producing mutation may be in the noncoding portions of PLP or in a nearby gene.

Adult↗
PubMed1996

Botulinum toxin in severe upper extremity spasticity among patients with traumatic brain injury: an open-labeled trial.

We studied the effect of botulinum toxin A (BTXA) among patients with traumatic brain injury (TBI) and severe spasticity unresponsive to conservative management. Twenty-one consecutive adult patients with severe spasticity involving the wrist and finger flexor musculature were treated with BTXA injection (20 to 40 units per muscle) under EMG guidance. After injection, patients received passive range of motion (ROM) exercise, with modalities and casting as clinically indicated. Outcome measures, including wrist ROM and the modified Ashworth Scale (MAS), were assessed 2 to 4 weeks after injection. Among the respective acute and chronic groups, mean ROM improved 42.9 (p = 0.001) and 36.2 degrees (p < 0.001). Mean MAS rating improved 1.5 (p = 0.01) and 1.47 (p = 0.002) points. There were no significant adverse effects. BTXA, in conjunction with conventional modalities, significantly improves spasticity and ROM in the distal upper extremity musculature of patients with TBI.

Adult↗