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PubMed2006

The Spastic Paraplegia Rating Scale (SPRS): a reliable and valid measure of disease severity.

OBJECTIVE: To develop and evaluate a clinical Spastic Paraplegia Rating Scale (SPRS) to measure disease severity and progression. METHODS: A 13-item scale was designed to rate functional impairment occurring in pure forms of spastic paraplegia (SP). Additional symptoms constituting a complicated form of SP are recorded in an inventory. Two independent patient cohorts were evaluated in a two-step validation procedure. RESULTS: Application of SPRS requires less than 15 minutes and does not require any special equipment, so it is suitable for an outpatient setting. Interrater agreement of SPRS was high (intraclass correlation coefficient = 0.99). Reliability was further supported by high internal consistency (Cronbach alpha = 0.91). SPRS values were almost normally distributed without apparent floor or ceiling effect. Construct validity was shown by high correlation of SPRS to Barthel Index and the International Cooperative Ataxia Rating Scale (convergent validity) and low correlation to Mini-Mental Status Examination (discriminant validity). CONCLUSION: The Spastic Paraplegia Rating Scale is a reliable and valid measure of disease severity.

Adolescent↗
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↗
PubMed1997

Homozygosity for an allele carrying intermediate CAG repeats in the dentatorubral-pallidoluysian atrophy (DRPLA) gene results in spastic paraplegia.

We report a family with autosomal recessive spastic paraplegia. Patient 1 was a 37-year-old woman and patient 2 was her 35-year-old sister. They showed spastic paraplegia with mild truncal ataxia and dysarthria but no dementia, epilepsy, myoclonus, or other involuntary movements. They were the products of a consanguineous marriage but the parents were neurologically normal. We analyzed the CAG repeats of the dentatorubral-pallidoluysian atrophy (DRPLA) gene in the family members. The patients were homozygous for an allele carrying an intermediate size of CAG repeats (41 or 40 repeats) in the DRPLA gene; the parents were heterozygous for an intermediate allele and a normal allele in this gene. Homozygosity for an intermediate allele in the DRPLA gene appears to have resulted in spastic paraplegia different from any DRPLA phenotype.

Adult↗
PubMed1999

Autosomal dominant spastic paraplegia: refined SPG8 locus and additional genetic heterogeneity.

OBJECTIVE: To map the gene responsible for autosomal dominant pure hereditary spastic paraplegia (ADPHSP) in a large affected family. BACKGROUND: Autosomal dominant pure hereditary spastic paraplegia (ADPHSP) is genetically heterogeneous, and loci have been mapped at chromosomes 2p (SPG4), 14q (SPG3), 15q (SPG6), and recently, in a single family, at chromosome 8q24 (SPG8). METHODS: The authors carried out a genomewide linkage screen on a large family with ADPHSP, for which linkage to the chromosome 2, 14, and 15 loci was excluded. RESULTS: Analysis of markers on chromosome 8q24 gave a peak two-point lod score of 4.49 at marker D8S1799. Analysis of recombination events in this family and in the previously published SPG8-linked family narrowed the SPG8 locus from 6.2 cM to a 3.4-cM region between markers D8S1804 and D8S1179. In another four families, linkage to all four known ADPHSP loci was excluded. The SPG8-linked family had a significantly older mean age at onset of symptoms and had significantly more wheelchair-using patients than the four linkage-excluded families. CONCLUSIONS: These results contain the presence of an autosomal dominant pure hereditary spastic paraplegia (ADPHSP) locus at chromosome 8q24 and strongly suggest that there are at least five ADPHSP loci. The data provide additional evidence for locus-phenotype correlations in ADPHSP.

Adult↗
PubMed2001

Dopa-responsive dystonia simulating spastic paraplegia due to tyrosine hydroxylase (TH) gene mutations.

Spastic paraplegia is not widely recognized to occur in dopa-responsive dystonia (DRD). The authors found a compound heterozygote for novel mutations of the human tyrosine hydroxylase (TH) gene (TH). The patient was initially diagnosed as having spastic paraplegia, but responded completely to levodopa therapy. Exercise-induced stiffness in the patient's father, who had a TH deletion, also responded to levodopa. The data expand the clinical spectrum of TH deficiency and suggest that TH mutations may account for some patients with DRD simulating spastic paraplegia.

Child↗
PubMed2000

Catecholamine response to exercise in individuals with different levels of paraplegia.

The purpose of this study was to investigate the effect of the level of injury on the serum level of norepinephrine (Nor) and epinephrine (Epi) at rest and after maximal exercise in individuals with paraplegia. Twenty-six male spinal cord-injured subjects with complete paraplegia for at least 9 months were divided into two groups of 13 subjects each according to the level of injury, i.e., T1-T6 and T7-T12. Serum Nor and Epi concentrations were measured by HPLC-ECD, at rest (PRE) and immediately after a maximal ergospirometric test (POST). Statistical analysis was performed using parametric and non-parametric tests. Maximal heart rate, peak oxygen uptake, and PRE and POST Nor were lower in the T1-T6 than in the T7-T12 group (166 +/- 28 vs 188 +/- 10 bpm; 18.0 +/- 6.0 vs 25.8 +/- 4.1 ml kg(-1) min(-1); 0.54 +/- 0.26 vs 0.99 +/- 0.47 nM; 1.48 +/- 1. 65 vs 3.07 +/- 1.44 nM). Both groups presented a significant increase in Nor level after exercise, while only the T7-T12 group showed a significant increase in Epi after exercise (T1-T6: 0.98 +/- 0.72 vs 1.11 +/- 1.19 nM; T7-T12: 1.24 +/- 1.02 vs 1.89 +/- 1.57 nM). These data show that individuals with paraplegia above T6 have an attentuated catecholamine release at rest and response to exercise as compared to subjects with injuries below T6, which might prevent a better exercise performance in the former group.

Adolescent↗
PubMed2005

Postoperative recovery of complete sudden paraplegia due to lumbar schwannoma. Case report.

The authors present a rare case of acute complete paraplegia due to a lumbar schwannoma. The clinical presentation, magnetic resonance imaging features, and management strategy are discussed. A 29 year-old man presented with acute complete paraplegia and bladder and bowel incontinence. He had a history of stable back pain and a 4-year history of lower-extremity numbness bilaterally. Magnetic resonance imaging revealed an enhancing extraaxial mass filling the spinal canal over two segments below the conus medullaris. An L1-3 laminectomy was performed and the tumor was completely removed. Pathological evaluation showed features characteristic of a schwannoma. The patient regained almost complete motor function after 6 months. To the authors' knowledge, this is the first reported case of acute complete paraplegia secondary to lumbar schwannoma. Possible mechanisms of this occurrence are discussed.

Acute Disease↗
PubMed2006

The effect of adjuvant perfusion techniques on the incidence of paraplegia after repair of traumatic thoracic aortic transections.

OBJECTIVE: To analyze the effect of adjuvant perfusion techniques of the distal aorta on the outcome of traumatic thoracic aortic transections. PATIENTS AND METHODS: From 1973 to 2004, 72 patients (mean age, 39 years) with thoracic aortic transections arrived alive at the emergency department. Nineteen patients arrived in extremis and underwent emergency operations, 42 patients were stable and underwent diagnostic evaluation before surgery (4 patients experienced aortic rupture during evaluation), and 11 patients presented more than 24 hours after the accident. Sixteen patients died before aortic repair could be performed. Operative repair was possible in 53 patients (46 stable and 7 in extremis). Interposition graft was performed in 47 patients, and primary repair was performed in 6 patients. Morbidity, mortality, and paraplegia rate were analyzed. RESULTS: Patients in extremis had a mortality rate of 84% (16 of 19), stable patients had a mortality rate of 11% (4 of 38), patients who experienced rupture during evaluation had a mortality rate of 100% (4 of 4), and patients who underwent delayed operation had a mortality rate of 0% (0 of 11). The paraplegia rate with and without adjuvant distal aortic perfusion techniques was 2% (1 of 41 patients) and 33% (4 of 12 patients), respectively (P=.007). Mortality and paraplegia rates were 4% and 4% for partial bypass (n=24), 42% and 33% for the clamp and sew technique (n=12), 0% and 0% for Gott shunt (n=10), and 29% and 0% for full cardiopulmonary bypass (n=7), respectively. CONCLUSIONS: Although thoracic aortic transections remain a highly lethal injury, hemodynamically stable patients have a low operative mortality. Spinal cord injury is decreased by the use of adjuvant perfusion techniques that maintain distal aortic perfusion during cross-clamping of the aorta.

Adult↗
PubMed1999

[Pott's disease paraplegia in children. Mechanics and therapeutic strategies. Six cases].

PURPOSE: We report a series of 6 Pott's disease paraplegias treated between 1982 and 1996. MATERIALS AND METHOD: Out of 15 children suffering from Pott's disease, 6 had paraplegia. Treatment consisted of anterior medullar decompression and anterior spine fusion with bone grafting. Two or three weeks later, posterior spine fusion was achieved systematically using a CD fixation device in 3 cases. RESULTS: Neurological signs completely disappeared in 5 children. Vertebral fusion was correct in all patients and kyphosis was less than 50 degrees. DISCUSSION: The posterior approach to the spine must be proscribed as a first step, except for spine dislocation. The anterior approach allowed us to drain the abscess, to correct the kyphosis, and to perform an anterior spine fusion. The posterior spine fusion was performed a few weeks later in order to avoid kyphosis aggravation. CONCLUSION: Prognosis of Pott's disease is good but at the present time, paraplegia remains too frequent. Adapted treatment must be performed without delay.

Age Factors↗
PubMed1999

Transient paraplegia following elective infrarenal aortic aneurysm repair. Case report.

Paraplegia is a well known complication after surgery for thoracic and thoraco-abdominal aneurysm but is very rare when the level involved is lower than the renal arteries. It is seen most often after treatment of ruptured aneurysm and very few cases are found in the literature reporting spinal cord ischemia after elective repair of an infrarenal abdominal aortic aneurysm. A new case of transient paraplegia following elective repair of an infrarenal abdominal aortic aneurysm is reported and different aspects of this complication are discussed. In our case, probably the interruption of blood flow in lumbar arteries and the duration of crossclamping were likely contributive factors and it suggest that a failure to appreciate the significance of collateral sources of spinal cord blood flow may be responsible for at least some cases of postoperative paraplegia.

Aged↗
PubMed1975

Spinal lesions, paraplegia and the surgeon.

Thirty-six patients with spinal cord lesions and varying degrees of paraplegia were seen by the surgical team at the Angau Memorial Hospital, Lae, over a thirty month period. Because the continued presence of a spinal lesion may lead to progressive cord destruction and ischaemic myelopathy, prompt treatment is advocated. The depressing results that have followed treatment of fracture dislocations of the cervical spine and secondary neoplasm with paraplegia is recorded and some suggestions are made that may improve the outlook in future cases. Early and major surgery is advocated in the treatment of spinal abscesses, tumours, Pott's paraplegia and unstable fracture dislocations of the lumbar spine.

Abscess↗
PubMed2000

Familial spastic paraplegia and maculopathy with juxtafoveolar retinal telangiectasis and subretinal neovascularization.

PURPOSE: To describe a previously unreported condition involving familial spastic paraplegia and a peculiar type of maculopathy. METHODS: Two pairs of siblings were studied. All four cases underwent serial clinical examinations, fundus photography, and fluorescein angiography. Two siblings had extensive investigations. RESULTS: Characteristics of the four cases include spastic paraplegia diagnosed in the first decade of life and visual loss at about age 9 due to a maculopathy with an abnormal vascular complex. In the early stages, parafoveal dilatation of the capillary network was noted. The later stages were characterized by cystic macular degeneration, and seven of eight eyes developed fibrovascular scars with retinochoroidal anastomoses, pigment migration, and atrophic changes. In two siblings, electro-oculographic findings were subnormal, whereas results of electroretinography, magnetic resonance imaging of the brain and spinal cord, and metabolic and karyotype studies were normal. These siblings were an Indonesian girl and boy; the other siblings were white males. There was no consanguinity of the parents and family history was unremarkable. CONCLUSIONS: This study suggests that the two pairs of siblings have an identical familial and probably recessive disorder with neurodegenerative changes that have caused paraplegia and a peculiar maculopathy associated with anomalous retinal vascular complexes, retinochoroidal anastomoses, and subretinal neovascularization.

Adolescent↗
PubMed1998

[Treatment of thoracolumbar tuberculosis complicated with paraplegia].

OBJECTIVE: To explore characteristics of thoracolumbar tuberculosis complicated with paraplegia, relationship between efficacy and approach of operation, and to discuss indication and applicability of conservative treatment. METHOD: Forty-eight cases with thoracolumbar tuberculosis complicated with paraplegia were classified into two groups. Thirty-two cases in the operational group were treated with extrapleural, extraperitoneal approach and transpleural, extraperitoneal approach. Sixteen cases in the nonoperational group were treated conservatively. RESULT: The recovered cases in the operational and the nonoperational groups respectively accounted for 75% and 88%. CONCLUSION: Thoracolumbar tuberculosis complicated with paraplegia can be cured by either operative or conservative treatment, and the efficacy of operation directly related with operational approach.

Adolescent↗
PubMed2002

[Non-traumatic paraplegia at the campus teaching hospital of Lome. Report of 243 cases].

To ascertain the aetiology, of non traumatic paraplegia retrospective survey, concerning 2396 patients hospitalised from January 1st 1995 to December 31st 1999, was done. Two hundred forty three cases were founded after investigation. The frequency of non traumatic paraplegia was 10.14%. Peripheral motor neuron's causes were 48.97% (119 cases). In this group, tropical neuromyelopathies were the commonest 65.54% (78 cases) while Guillain Barre syndrome was 3.36% (4 cases). Bones, epidural et meningeal (envelope) causes were 38.68% (94 cases). In this group, tuberculosis was 17 cases, tumour 36 cases. Central motor neuron causes were 9.46% (23 cases) with HIV myelitis 13 cases, tumour 3 cases, degenerative conditions 6 cases. It is concluded that the major causes of non traumatic paraplegia in Lome are similar to that reported from sub sahara countries with tropical neuromyelopathies, tumours, tuberculosis and HIV myelitis.

Adolescent↗
PubMed2002

Tuberculous paraplegia in pregnancy treated by surgery.

Tuberculous paraplegia in pregnancy is reported to be rare. Paraplegia due to tuberculosis has a good prognosis if surgical decompression and stabilisation are done early together with chemotherapy. Vaginal delivery is not contraindicated in pregnancy complicated by paraplegia, but is associated with problems related to the initiation and progression of labour. Performing spinal nursing on an unstable spine with a rapidly enlarging gravid uterus in the third trimester of pregnancy poses a significant challenge. We report successful simultaneous Caesarean section and surgical treatment of a paraplegic spine due to tuberculosis.

Adult↗
PubMed2002

[A case of SAPHO syndrome with paraplegia due to a thoracic kyphosis].

A 63-year-old man visited our hospital in January 1993 because of back pain, which had been present for a year and persisted. The patient was diagnosed compression fracture of thoracic spine by another hospital. Thoracic plain radiographs revealed destructive and sclerotic changes with reduction of height of T 8, T 9 vertebral body. He had kyphosis on this level. Radiographs of the chest revealed hyperostosis of bilateral proximal clavicle. We diagnosed SAPHO syndrome (synovitis, acne, pustlosis, hyperostosis, and osteomyelitis: SAPHO) with T 8, T 9 spondylodiscitis, however without any skin manifestations. Oral indomethacin was effective, however thoracic kyphosis progressed gradually. Spastic gait and paraplegia appeared from February 1998, at last on July he was unable to walk independently. MRI showed the compression of spinal cord on T 8, T 9 level. We performed circumferential decompression and fusion with instrumentation. His paraplegia improved after surgery. We describe a rare case of SAPHO syndrome with paraplegia due to a thoracic kyphosis.

Acquired Hyperostosis Syndrome↗
PubMed1992

The effect of an anti-G suit on cardiovascular responses to exercise in persons with paraplegia.

The purpose of this study was to determine whether external pressure on legs and abdomen could prevent venous blood pooling in persons with paraplegia and thus positively affect their cardiovascular responses to arm exercise. To investigate this, five male subjects with paraplegia (P), with complete lesions between T6 and T12, and five male control subjects who were wheelchair bound (C) (due to a chronic lower extremity disability), performed submaximal arm-cranking exercise at 20%, 40%, and 60% of their maximal power output (Wmax), with and without an antigravity (anti-G) suit inflated to 52 mm Hg (1 psi). For P, higher preexercise systolic pressure (127 vs 117 mm Hg) was seen with the anti-G suit. At 40 and 60% Wmax, significantly lower heart rates (at 40% = 5.7%; at 60% = 10.6%) at similar cardiac outputs were seen for P with an anti-G suit. Although not significant, P also demonstrated higher stroke volumes at 40% (4.8%) and 60% (5.0%) Wmax with external pressure. For C, no differences in preexercise blood pressure or cardiovascular responses at all three exercise levels were seen with or without the anti-G suit. These data suggest that an inflated anti-G suit is able to prevent venous blood pooling and offers hemodynamic benefits in persons with paraplegia during submaximal arm-cranking exercise. In addition, this study reports a possible alternative to hosiery or functional neuromuscular stimulation that could be applied to all subjects with spinal cord injuries regardless of type or duration of the lesion or of muscle-atrophy.

Adult↗
PubMed2004

Clinical features of hereditary spastic paraplegia with thin corpus callosum: report of 5 Chinese cases.

BACKGROUND: Hereditary spastic paraplegia is a clinically and genetically heterogeneous group of neurodegenerative disorders of the motor system, characterized by slowly progressive spasticity and weakness of the lower extremities. This study was conducted to investigate the clinical features of hereditary spastic paraplegia with thin corpus callosum (HSP-TCC). METHODS: Clinical data from five patients and thirty-five previously published case reports of HSP-TCC were analyzed retrospectively. RESULTS: Most patients were adolescents at the onset of the disease, presenting with spastic paraparesis of the lower limbs and mental impairment. Some patients also had other clinical features, including spasticity of the upper limbs, cerebellar ataxia, and sensory disturbances. Cranial MRIs of the five patients revealed an extremely thin corpus callosum, sometimes with widened cerebral sulci and ventricles, as well as with cerebellar and cerebral atrophy. CONCLUSION: The main clinical features of HSP-TCC include slowly progressive spastic paraplegia, mental impairment during the second decade of life, and an extremely thin corpus callosum as shown on cranial MRIs.

Adolescent↗