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Two patients are reported with Ekbom's syndrome of "restless legs" occurring in association with arborizing telangiectasia of the lower limbs. Sensory complaints have previously been reported in this skin condition but not described in detail. The co-existence of the two conditions is discussed in the context of previous explanations of the restless legs syndrome.
This study measured oxygen uptake (VO2), minute ventilation (VE), and heart rate (HR) in a bilateral above-knee (AK) amputee and in three able-bodied controls during progressive treadmill exercise. Walking conditions for the amputee included using bilateral short-leg (SL) and long-leg (LL) prostheses. A progressive treadmill protocol to maximal capacity was used for the amputee and duplicated by the control subjects. An automated system was used to measure VO2, VE, and HR throughout exercise. Data analysis was restricted to the use of parameter averages and percentages to describe differences between experimental conditions. Maximal VO2 for the amputee averaged 23.3mL/kg-1/min-1 with the LL and 22.8mL/kg-1/min-1 with the SL prostheses, a negligible difference between conditions; however, exercise duration was 27% longer when using the SL prostheses. In addition, when averaged over the first four stages of exercise, VO2, VE, and HR were 24%, 32%, and 14% higher, respectively, when the LL prostheses were used. Treadmill walking by unimpaired controls averaged 47% and 79% more economical than walking with the SL or LL prostheses, respectively. These results demonstrate that the use of currently available AK prostheses requires significant energy expenditure, which limits their use to only the most physically fit individuals.
Neurons within the precursor of the adult leg, the imaginal disc, innervate a larval sense organ, Keilin's organ. Electron microscopical investigations of first instar larvae show that five dendrites end at the organ: three insert at the bases of the three hairs of the organ and two end against the cuticle, without any apparent cuticular specialization. In third instar larvae, the imaginal leg discs invaginate into the body cavity, and only four of the dendrites (the outer segments of which become greatly elongated) remain in contact with Keilin's organ. The axons of the neurons that supply Keilin's organ project into a ventral neuropile region of the central nervous system, with a pattern that resembles the projections of other larval sensilla. Electrical activity can be recorded from neurons of the imaginal disc in response to mechanical stimulation.
Attenuation of initial somatosensory evoked potential (SEP) gain becomes more pronounced with increased rates of movement. Manipulation of the range of movement also might alter the SEP gain. It could alter joint receptor discharge; it should alter the discharge of muscle stretch receptors. We hypothesized that: (1) SEP gain reduction correlates with both the range and the rate of movement, and (2) manipulation of range and rate of movement to achieve similar estimated rates of stretch of a leg extensor muscle group (the vasti) results in similar decreases in SEP gain. SEPs from Cz', referenced to Fpz' (2 cm caudal to Cz and Fpz, respectively, according to the International 10-20 System), along with soleus H-reflexes were elicited by electrical stimulation of the tibial nerve at the popliteal fossa. Stable magnitudes of small M-waves indicated stability of stimulation. A modified cycle ergometer with an adjustable pedal crank and electric motor was used to passively rotate the right leg over three ranges (producing estimated vasti stretch of 12, 24 and 48 mm) and four rates (0, 20, 40 and 80 rpm) of movement. Two experiments were conducted. Ranges and rates of pedalling movement were combined to produce two or three equivalent estimated rates of tissue stretch of the vasti muscles at each of 4, 16, 32 and 64 mm/s. Tibial nerve stimuli were delivered when the knee was moved through its most flexed position and the hip was nearing its most flexed position. Means of SEP, H-reflex and M-wave magnitudes were tested for rate and range effects (ANOVA). A priori contrasts compared means produced by equivalent estimated rates of vasti stretch. Increasing the rate of movement significantly increased the attenuation of SEP and H-reflex gain (P<0.05). Increasing the range of movement also significantly increased these gain attenuations (P<0.05). Combining these to achieve equivalent rates of stretch, through different combinations of rate and range, resulted in equivalent depressions of SEP gain. H-reflex gains were similarly conditioned. These results suggest that muscle stretch receptors play a more important role than joint or cutaneous receptors in regulating SEP gain consequent to movement. We note that the present calculation only considers the knee extensors; however, the biomechanical model of stretch applies also to receptors in the hip extensors. This paper and the companion one show that primary factors in the kinaesthetic components of the movement regulate activity-induced gain attenuation of SEPs.
The restless legs syndrome (RLS) is a clinical diagnosis based on the four essential criteria defined by the International Restless Legs Syndrome Study Group (IRLSSG). An idiopathic form can be separated from a symptomatic form. Neurophysiological studies have investigated the pathophysiology of the idiopathic RLS or have been used to exclude a symptomatic cause, in particular polyneuropathy. So far cortical excitability changes, corticomotor, somatosensory and auditory pathways, spinal cord excitability, B-wave rhythm and cycling alternating pattern, as well as reflex mechanisms have been investigated by electroencephalography, evoked potentials, Bereitschaftspotentials, nerve conduction and thermal threshold measurements, electromyography, transcranial Doppler sonography, measurements of the spinal flexor reflex as well as neuroimaging techniques. The etiology of the RLS cannot be revealed by these methods, neurophysiological studies in RLS are, however, useful for a better understanding of the pathophysiology and for exclusion of a polyneuropathy or other symptomatic causes. In addition to neurophysiological investigations, small fiber neuropathy, which seems to be a more common finding in RLS patients than expected to date, may need biopsy for confirmation. This review will focus on investigations of the different systems involved with diverse neurophysiological methods.
The Ilizarov technique has been used to treat severe limb length discrepancy and short stature. However, complications of this treatment are frequent. Between 1984 and 2001, 57 patients (94 tibias) had an Ilizarov procedure for limb lengthening. Twenty patients had limb discrepancy and 37 had short stature. Their mean age was 20.2 years (range 15-34). The average limb lengthening was 8.37 cm (range 3.2-14.7), which was equivalent to 26% (range 9.2-60%) average tibial lengthening. A total of 90 complications were observed. Thirty-three unplanned procedures were required during the lengthening programme. Two patients stopped the lengthening programme. There was no difference in the complications in leg lengthening using Ilizarov technique between the group of patients with leg length discrepancy and the group with short stature. A good knowledge of the Ilizarov technique is necessary to perform a lengthening programme with a low rate of complications.
In a series of 165 cross-leg flaps, 94 per cent were successful. Some degree of necrosis occurred in almost half of them. Major necrosis requiring split-skin grafting occurred in less than one-fifth. Thrombo-embolic complications were rare and mainly in the older age group. The retunr of sensation was good; better in the flap than in the donor site. The long-term results were good with a low complication rate. On weight-bearing surfaces the cross-leg flap skin stands up well to wear and tear.
OBJECTIVE: Periodic leg movements during sleep (PLMS) are often associated with electroencephalographic (EEG) changes, such as microarousals (MA), and with heart rate (HR) variations. The aim of the present study was to evaluate the effects of age and gender on HR changes associated with PLMS in restless legs syndrome (RLS) patients. METHODS: Forty-two RLS patients underwent one night of polysomnographic recordings. They were divided into 3 groups of 14 subjects (7 women and 7 men) according to age, i.e. young (25-40 years), middle-aged (41-55 years) and elderly (56-71 years) patients. The RR interval was calculated for 5 intervals before and 15 intervals after the onset of 50 PLMS in each patient. RESULTS: PLMS were associated with HR changes characterized by a tachycardia followed by a bradycardia. However, a reduction in the tachycardia and the bradycardia was observed with age. Moreover, women showed a higher amplitude in the bradycardia than men. No age or gender difference was found for MA index and duration. CONCLUSIONS: This study showed age and gender differences in the magnitude of the HR changes associated with PLMS. The knowledge of HR variations during sleep, including rapid HR changes associated with sleep events such as PLMS or MA, may be helpful in understanding the potential mechanisms involved in the increased cardiac risk observed in elderly.
UNLABELLED: Two separate studies are described using the same prostacyclin analogue in a similar group of patients. OBJECTIVES: to assess the tolerability and efficacy of two dose regimens of oral Iloprost compared with placebo in the treatment of patients with ischaemic ulcers, gangrene or rest pain due to severe arterial disease over a period of 4 weeks (Study A) and one year (Study B). DESIGN: multicentre, placebo controlled, double-blind, randomized prospective studies. SUBJECTS & METHODS: 178 (study A) and 624 (study B) patients with trophic skin lesions (ulcers or gangrene) or ischaemic rest pain due to severe arterial disease. To confirm severe arterial disease patients were required to have a systolic ankle Doppler pressure of 70 mmHg or less or a toe systolic Doppler pressure of 50 mmHg or less in one leg. In both studies patients were randomly allocated to three treatment groups: placebo, low dose Iloprost (50-100 microgram twice a day) or high dose (150-200 microgram twice a day) In Study A the main outcome measures were tolerability of different doses of Iloprost and death, major amputation, healing of trophic lesions and relief of rest pain at the end of the follow up, which was 5 months after the end of the treatment. In Study B the primary end point was time to major amputation and stroke or death up to 12 months. Secondary pre-defined end points included the combined end point of patients alive without amputation, no trophic skin changes, no rest pain and not on regular analgesics. RESULTS: the proportion of patients who completed the 4-week treatment period in Study A at the intended dose was 58%, 43%, 45% respectively in the placebo, low dose and high dose Iloprost groups. In an intention to treat analysis the proportion of patients who survived without major amputation, ulcers or gangrene and had no rest pain was 11% in the placebo group, 19% in the low dose iloprost group and 28% in the high dose Iloprost group. The pooled Iloprost groups showed a statistically significantly better result than the placebo group (p=0.04), as did the high dose Iloprost group compared to the placebo (p=0.014). In Study B there was no treatment benefit in terms of a primary end point of amputation and death. However the secondary combined end point of patients who survived without a major amputation, ulcers or gangrene and had no rest pain, nor a need for regular analgesia was favourable for Iloprost, with 18% of patients in the placebo group reaching this optimal secondary end point, compared to 23% in the low dose Iloprost group and 26% in the higher dose Iloprost group (p<0.05). CONCLUSIONS: oral Iloprost administered for a year showed no clear benefit in patients with advanced severe leg ischaemia (PAOD III and IV). The results obtained with 4 weeks' treatment in Study A and in previous trials of intravenous Iloprost could not be reproduced
The distraction-epiphyseolysis is a method of leg-lengthening and allows to correct leg-deformities. A daily lengthening of 1 mm enhances ossification and enables a maximum lengthening of 8-10 cm. The indications, contra-indications and the method are described.
BACKGROUND: In a clinical study evaluating the functioning of mobility-limited elders, muscle power accounted for more of the variation in function than strength did. There was also evidence that the power-function relationship may be described as curvilinear. However, these findings have never been confirmed in a representative population. METHODS: An analysis was conducted using data from the InCHIANTI study, a population-based cohort of 1032 adults living in Italy. To assess the relationships between impairments in power and strength and mobility performance, we created separate multivariate linear and log-transformed regression models as well as separate logistic regression models. RESULTS: Subjects were age > or = 65 years, predominantly female (54%), with a mean age of 74.2 years, and most had mild-moderate mobility limitations (Short Physical Performance Battery score 10.5 +/- 2.1). Though leg extension power and isometric hip extension strength were closely associated, in several separate multivariate linear regression models, leg power consistently explained more of the variance than strength did in several measures of physical performance. Differences were even larger when observed in curvilinear models of power and strength. Using separate multivariate logistic regression models to examine the odds ratios for mobility limitations in persons with low muscle power versus high muscle power, and in persons with low muscle strength versus high muscle strength, we found that both factors influenced risk for mobility limitations, but low power was associated with a 2-3-fold greater likelihood than low strength. CONCLUSION: These findings identified muscle power to be a more influential proximal determinant of physical performance than impairments in strength and emphasized muscle power as an important determinant of mobility skills in older adults.
STUDY OBJECTIVES: Polysomnographic study evaluating the efficacy of ropinirole for the treatment of patients with restless legs syndrome (RLS) suffering from periodic leg movements in sleep (PLMS). DESIGN: Double-blinded, placebo-controlled, parallel-group study. SETTING: 15 tertiary referral centers in the USA. PARTICIPANTS: 65 patients with RLS and PLMS. INTERVENTIONS: Ropinirole (0.25-4.0 mg per day) or placebo for 12 weeks. MEASUREMENTS AND RESULTS: Data from 59 patients were included in the primary endpoint analysis. PLMS per hour decreased more with ropinirole (48.5 to 11.8), compared with placebo (35.7 to 34.2; adjusted treatment difference: -27.2; 95% confidence interval [CI]: -39.1, -15.4; P < .0001). Periodic limb movements with arousal per hour decreased from 7.0 to 2.5 with ropinirole but increased from 4.2 to 6.0 with placebo (adjusted treatment difference: -4.3, 95% CI: -7.6, -1.1; P = .0096). Periodic limb movements while awake per hour decreased from 56.5 to 23.6 with ropinirole but increased from 46.6 to 56.1 with placebo (adjusted treatment difference: -39.5; 95% CI: -56.9, -22.1; P < .0001). Ropinirole treatment significantly improved patients' ability to initiate sleep (P < .05) and the amount of Stage 2 sleep compared with placebo (P < .001). There were also non-significant trends toward increases in total sleep time and sleep efficiency. Sleep adequacy (measured on the subjective Medical Outcomes Study sleep scale) was significantly improved with ropinirole treatment (adjusted treatment difference: 12.1; 95% CI: 1.1, 23.1; P = .0316). In contrast, the placebo group showed a greater increase in Stage 3/4 sleep (P < .01). No serious adverse events occurred in either group. CONCLUSIONS: Ropinirole is effective in the treatment of both the sleep and waking symptoms of RLS.
Dopaminergic treatment is very effective in restless legs syndrome (RLS) and periodic leg movements in sleep (PLMS). However, neuroreceptor imaging studies that addressed altered striatal dopaminergic function have given controversial results. In this present study, 14 patients with idiopathic RLS (iRLS) and PLMS with a good response to dopaminergic and non-dopaminergic treatment and ten healthy sex- and age-matched controls were investigated off-medication by using 123I-IBZM and SPECT. RLS symptoms and sleep disturbances were evaluated using three nights of polysomnography, the Pittsburgh Sleep Quality Index, and the International RLS Study Group (IRLSSG) rating scale. The patients presented with sleep disturbances, a high PLMS index (56.2 +/- 33.1 per h), and severe RLS symptoms during SPECT (IRLSSG rating scale 23.1 +/- 8.0), and showed no significant differences in striatal to frontal IBZM binding to D2 receptors compared to controls (ratio striatum/frontal cortex, right side 1.60 +/- 0.10 vs 1.63 +/- 0.08, P = 0.35, NS; left side 1.61 +/- 0.11 vs 1.63 +/- 0.08, P = 0.51, NS). These findings show normal function of striatal D2 receptors in successfully treated patients with iRLS and PLMS. Dopaminergic and non-dopaminergic pretreatment does not appear to change striatal D2 receptor binding as compared to healthy controls. Structures other than striatal D2 receptors are discussed as possible causes of the treatment effects in RLS.
Tissue expansion of adjacent intact skin and subcutaneous tissue has in five legs provided high-quality soft-tissue coverage of the distal end in legs amputated at different levels without further shortening of the bone. The sensitivity of slowly expanded flaps is temporarily affected but eventually seems to return to normal. Expanded flaps have endured both sitting and the use of prostheses for 4 to 5 years. In one case the technique made it possible to save the knee joint. Besides being a functional reconstruction, the appearance of the reconstructed parts much improved. A technique of overexpansion and double flap coverage of the bone utilizing deepithelialization is described.
OBJECTIVE: To assess long-term outcome and prognostic factors for extreme surgery by vascular and plastic surgical teamwork for leg salvage in patients with critically ischemic large tissue defects. SUMMARY BACKGROUND DATA: Combined vascular reconstruction and microvascular free-flap transfer has been used to improve distal perfusion and cover large tissue defects caused by the critical limb ischemia (CLI) in few dedicated centers during the past 15 years. Comorbidities compromise the results of these demanding operations, and it is unclear how far this mode of treatment should be extended. METHODS: During 1989 to 2003, altogether 2157 vascular or endovascular revascularizations for CLI manifested as tissue lesions were performed. These included 81 revascularizations combined with microvascular free flap transfers in 79 patients (37-85 years). All the patients were candidates for major amputation. The patients were followed up at least 2 years or to death (mean follow-up, 62 months; SD, +/-34 months). RESULTS: One- and 5-year leg salvage rates were 73% and 66%, survival rates 91% and 63%, and amputation-free survival rates of 70% and 41%, respectively. Male gender and American Society of Anesthesiologists score 4 were associated with an increased risk of death, whereas the involvement of the heel mostly with calcaneal osteomyelitis and a large size of defect predicted major amputation. CONCLUSIONS: A combined vascular reconstruction and free-flap transfer offers an option for advanced limb salvage in a selected group of patients with CLI and a major tissue defect. Poor general condition, the involvement of the heel, and a large defect would indicate an amputation over extreme attempts for limb salvage.
The blood flow rate in subcutaneous adipose tissue was measured on the lower legs of 11 patients with chronic lower-leg venous insufficiency and ulceration and in eight age-matched control subjects for 12-20 h, under ambulatory conditions, using the 133Xe wash-out technique with portable Cadmium telluride (CdTe(Cl)) detectors. In both groups, the change from an upright to a supine position at the beginning of the night period elicited an instantaneous increment in the blood flow rate of 30-40% with a decrease in the central and local postural sympathetic vasoconstrictor activity. After approximately 1 h of sleep, a considerable increase in blood flow rate was seen in both patient and control groups which persisted for nearly 100 min. In the patient group, the mean increase was 137% compared to a mean increase of 68% in the control group (P less than 0.01). The blood flow then returned to the same level as at the beginning of the night and remained stable until the subjects woke in the morning. The differences between this nocturnal hyperaemic phase and the adjacent phases were highly significant in both groups (P less than 0.0001 and P less than 0.005, respectively). The blood flow levels measured on the second day were of the same magnitude as those registered on the first day. The mean values of the wash-out rate constants (k) of the two groups were of the same magnitude in all phases of subcutaneous blood flow level, except in the hyperaemic phase, where it was significantly higher in the patient group (P less than 0.01).(ABSTRACT TRUNCATED AT 250 WORDS)
The restless legs syndrome (RLS) is a common sensorimotor disorder which leads to severe sleep disturbances and showed a prevalence of 7.9% in our sleep laboratory. The aim of this study was to investigate periodic leg movements (PLM), arousal and respiratory variables in 12 untreated RLS patients and to measure the acute effects of 0.5 mg ropinirole, a nonergoline dopamine agonist, as compared with placebo. In the target variable PLM/h of total sleep time (PLM/h TST), RLS patients showed an increased value of 40/h (normal 0-5/h). Further, we found an increased number of PLM (368), PLM/h of time in bed (49/h), PLM/h of REM sleep (11), PLM/h of non-REM sleep (46) and PLM/h awake (61). The arousal index was also increased (32/h; normal 0-25/h), as were arousals due to PLM. In the confirmatory part of our descriptive data analysis, ropinirole 0.5 mg significantly improved, as compared with placebo, the index PLM/h TST by 75%. In the descriptive part, all the other PLM variables were improved as well. Arousals due to PLM decreased, while spontaneous arousals increased. Respiratory variables, which had a priori been in the normal range, showed a slight but significant improvement after the dopamine agonist. Thus, 0.5 mg ropinirole significantly improved the target variable PLM/h TST, along with objective and subjective sleep quality and morning noopsychic performance, as described in the preceding paper. Our data encourage further sleep studies including all above-mentioned variables in a larger group of RLS/PLM during sleep patients as well as long-term efficacy trials.
We report here the possible effect of opiates on a patient exhibiting particularly severe restless legs syndrome (RLS) and periodic leg movements in sleep (PLMS). This patient was investigated in the sleep laboratory under three conditions, namely, unmedicated (baseline), medicated with codeine sulfate, and medicated with both codeine sulfate and pimozide. Codeine sulfate dramatically improved abnormal motor behavior in this patient. The addition of pimozide reversed the beneficial effect of codeine during the Forced Immobilization Test but not in spontaneous RLS or PLMS at night. These results are discussed in view of the possible involvement of the dopaminergic mechanism in RLS/PLMS syndrome.