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PubMed1980

Factors influencing dementia and epilepsy in Huntington's disease of early onset.

A sample of young patients with Huntington's disease, ascertained from case material reported in the literature, was examined to determine whether muscular rigidity or age at onset of the disorder is the major factor in giving rise to early dementia and epilepsy. Because of correlation between explanatory variables, adjustment for their effects was made in calculating the strength of associations. The risk of incurring dementia early was found to be over twice as great for rigid patients than for those without the sign. Onset age was independently involved with rigidity in this relationship. When the effect of muscular rigidity was adjusted for sex and onset age, virtually no influence on the occurrence of epilepsy was detected. Onset age, on the other hand, was strongly associated with the likelihood of epileptic attack.

Adolescent↗
PubMed2000

Forty-five years of stereotactic surgery for Parkinson's disease: a review.

An overview of a large experience of the surgical management of parkinsonism from 1954 to 1999 is outlined--from the original open surgery of Fenelon to the sophisticated, motor-driven electrodes of stereotactic surgery using depth microelectrode recording. The definition of the best target sites for the relief of tremor, rigidity and bradykinesia evolved progressively as accuracy in siting of lesions developed. The significance of these targets in understanding the pathophysiology of the disease and the neurotransmitters involved gradually became clearer and a way forward in future management was suggested.

History, 20th Century↗
PubMed1992

[Comparative evaluation of various forms of Parkinson disease in middle-aged and elderly patients (clinical, neuropsychological and computerized-tomographic studies)].

The clinical, neuropsychological and computer-aided tomography data were compared in elderly and senile patients suffering from trembling and akinetic rigid forms of parkinsonism. 12 patients with trembling and 21 with akinetic rigid parkinsonism were examined. The groups did not differ in age or sex. The results of computer-aided tomography of the brain were evaluated using linear, volumetric and densitometric parameters. The akinetic rigid pattern was found to be associated with a more diverse clinical and neuropsychological symptomatology, by more manifest cerebral atrophy. The gravity of the status of patients with trembling parkinsonism is largely determined by the intensity of the hyperkinetic syndrome, that of patients with akinetic rigid parkinsonism not only by amyostatic disturbances but also by the presence and severity of incoordination , pseudobulbar and mnemonic intellectual disorders. The disorders identified in patients with akinetic rigid parkinsonism often attest to dysfunction of the frontal parts of the brain. The nosological heterogeneity of both patterns of parkinsonism is assumed.

Age Factors↗
PubMed1992

[Comparative evaluation of various forms of parkinsonism in middle-aged and elderly patients (clinical, neuropsychological and computerized-tomographic studies)].

The clinical, neuropsychological and computed tomography data were compared in elderly and senile patients with trembling and akinetico-rigid forms of parkinsonism. 12 patients with trembling and 21 with akinetico-rigid forms were examined. The groups did not differ in age or sex. The results of computed brain tomography were estimated with the use of linear, volumetric and densitometric parameters. The akinetico-rigid form was associated with more diverse clinical and neuropsychological symptomatology, more noticeable cerebral atrophy. The gravity of the status of the patients with trembling parkinsonism was largely specified by the intensity of the hyperkinetic syndrome whereas that of the patients with the akinetico-rigid form not only by amyostatic disorders but also by the presence and severity of discoordination, pseudobulbar and mnemonic-intellectual disturbances. Disorders revealed in the patients with the akinetico-rigid form often attest to the dysfunction of the frontal parts of the brain. The nosological heterogeneity of these two forms of parkinsonism is under discussion.

Age Factors↗
PubMedDate not supplied

Prognosis of traumatic decerebrated rigidity.

91 cases with traumatic decerebrated rigidity are reported and compared with age of patients, kind of lesion, time of decerebration. The Authors consider the reversibility of some of these comas with decerebrated rigidity and try to establish some immediate criteria for prognosis, and, in the long term, the quality of the functional recovery achieved.

Adolescent↗
PubMedDate not supplied

Levetiracetam-induced parkinsonism in a Huntington disease patient.

The authors present a man with Huntington disease who was treated with levetiracetam (Keppra) in an effort to reduce chorea. Chorea was markedly reduced, but the patient developed parkinsonism and lethargy after 6 weeks of treatment. Symptoms consisted of resting tremor, rigidity, increased dystonia, and gait difficulty. Side effects from levetiracetam resolved completely within 7 days of levetiracetam discontinuation, and chorea returned to baseline.

Antipsychotic Agents↗
PubMed1980

Seven cases of Huntington's disease in childhood and levodopa induced improvement in the hypokinetic--rigid form.

Seven cases of the infantile form of Huntington's disease--six boys and one girl--were observed in the space of six years. Six children showed the hypokinetic-rigid form of the disease. Results of clinical, speech evaluations, laboratory tests, EEG and LPEG corresponded with data in literature. Initial signs and symptoms were mild sensorimotor and behavioural disturbances. In five patients, all affected by the hypokinetic-rigid form, CSF HVA and 5HIAA were determined. CSF HVA levels were found to be significantly lowered in three patients. One child showed significant decrease of CSF 5HIAA. Levodopa, orally administrated to these five patients during 8 days to 6 weeks in dosages of 75 to 600 mg per day, induced in all a marked improvement of the hypokinesia and/or rigidity, speech and social behaviour. Two children developed slight choreatic movements and one child had to be withdrawn from the drug because of decrease of appetite.

Adolescent↗
PubMed1995

Striatal D1 and D2 receptor binding in patients with Huntington's disease and other choreas. A PET study.

We have used PET to study striatal D1 and D2 receptor binding in 10 patients with either the choreic or akinetic-rigid variants of Huntington's disease and in three patients with other causes of chorea. Background rigidity and bradykinesia in choreic patients were scored with a four-point scale. PET studies showed a severe and parallel reduction of both striatal D1 and D2 receptor binding in Huntington's disease patients irrespective of their predominant phenotype (mean reduction 60%). Huntington's disease patients with rigidity showed more pronounced reduction of striatal D1 and D2 binding compared with those without rigidity. A case of chorea associated with systemic lupus erythematosus had normal D2 binding. These results suggest that the presence of chorea per se may not be determined by alterations in striatal dopamine receptor binding, but that rigidity in Huntington's disease is associated with severe striatal D1 and D2 receptor loss.

Adult↗
PubMed1996

Non-Parkinson akinetic-rigid syndromes.

We review developments in the study of akinetic-rigid syndromes other than Parkinson's disease within three main categories: recording of new etiologies, advancing knowledge on previously known causes, and studies of Parkinson's disease imitators and clinical and investigational means of accurately distinguishing them from Parkinson's disease.

Diagnosis, Differential↗
PubMed1997

Bilateral posteroventral pallidotomy in advanced Parkinson's disease in three patients.

In this report, we describe the effect of staged bilateral posteroventral pallidotomy in three patients with advanced Parkinson's disease who were all of the young-onset type. Two patients had developed response fluctuations after the use of levodopa, with severe hypokinesia, painful dystonia, and rigidity in the "off" phase and violent dyskinesias in the "on" phase. One patient, in a continuous hypokinetic rigid state, was totally unresponsive to dopaminergic medication. All were at Hoehn and Yahr stage 5 in the "off" phase before surgery. After surgery, the hypokinetic state was reversed and dyskinesias were abolished in all patients. Hoehn and Yahr stages were 3 in the "off" phase postoperatively. Overall functional improvement was marked and lasting after follow-up for 7, 12, and 13 months, respectively. Complications were visual field deficit and transient central facial paresis, both in the same patient. Bilateral posteroventral pallidotomy can ameliorate response fluctuations, hypokinesia, rigidity, and painful dystonia in advanced Parkinson's disease.

Activities of Daily Living↗
PubMed1999

Differential diagnosis of parkinsonism.

The diagnosis of Parkinson's disease is predominantly clinical, based on a combination of the cardinal features of tremor, bradykinesia and rigidity. The differential essentially lies between other conditions resulting in tremor, of which essential tremor is the commonest, and other akinetic-rigid syndromes. These include progressive supranuclear palsy, multiple system atrophy, toxins and other degenerative disorders, including diffuse Lewy body disease and corticobasal degeneration. The key clinical features of these disorders and a practical diagnostic approach are briefly reviewed in this article.

Diagnosis, Differential↗
PubMed1997

Truncal rigidity as a result of epidural sufentanil--a case report.

It is well known that intravenous opioids may cause truncal rigidity. To the best of our knowledge truncal rigidity induced by epidural opioid has never been reported. Recently, we came across an accident of truncal rigidity following epidural sufentanil. The victim was a 65-year-old female who received cholecystectomy, choledochotomy, and cholangiography. For post-operative pain control, an epidural catheter was inserted cephalad [corrected] at L1-2 interspace with a length of 4 cm of the catheter retained in the epidural space. The epidural catheter was secured and tested for correct placement with 3 ml of 2% lidocaine with 1:200,000 epinephrine prior to induction of general anesthesia. No opioid was ever given in the operative course. When the patient was fully awake and complained of wound pain in the recovery room 50 mg of sufentanil in 10 ml normal saline was given via the epidural catheter after a negative evacuation test. About one minute after the epidural shot, she was found to lose consciousness without any slightest warning sign. Truncal rigidity and locked jaw that followed entailed respiratory arrest and rapid deterioration of oxygenation which evidenced a life-threatening airway emergency. It spite of our efforts we could not manage to ventilate her with ordinary means. It was not until the administration of 80 mg of succinylcholine and oral endotracheal intubation could an adequate ventilation be reestablished. She regained spontaneous breathing 15 min after the episode but for safety's sake she remained intubated for 6 h until the dissipation of analgesia. Another test dose was attempted, which reconfirmed that the epidural catheter was in proper position. She stayed in the recovery room for 24 h and returned to ward in satisfactory condition. The incidence disclosed that epidural sufentanil even with a dose as small as 50 micrograms could cause truncal rigidity. Thus when epidural sufentanil is applied for post-operative pain control constant vigilance is necessary in order to avoid accident.

Abdomen↗
PubMed1998

Truncal muscle tonus in progressive supranuclear palsy.

OBJECTIVE: To elucidate the character and distribution of the abnormal muscle tonus in the body axis in progressive supranuclear palsy. Although neck hypertonus has been well described in progressive supranuclear palsy, little is known about the involvement of the truncal muscles. METHODS: Muscle tonus of the neck and trunk was separately investigated in 13 patients with progressive supranuclear palsy by clinical examination and surface EMG during passive rotation. Muscle hypertonus was graded according to a four point scale, and subjected to statistical analysis. The results were compared with those from 13 age matched patients with Parkinson's disease and six healthy volunteers. RESULTS: In all but one patient with progressive supranuclear palsy, there was a distinct difference in muscle tonus between the neck and trunk. A tonic shortening reaction characteristic of dystonia and an increased tonic stretch reflex (rigidity) were present in the neck muscles of patients with progressive supranuclear palsy, whereas only normal to moderately increased tonus was noted in the truncal muscles (neck v trunk, shortening reaction p=0.0001; stretch reflex p=0.0241). Follow up studies disclosed an increase in axial muscle tonus with predilection for the neck in three of four patients. In the 13 patients with Parkinson's disease, however, no significant difference was found in muscle rigidity between the neck and trunk. CONCLUSION: Mild changes in truncal muscle tonus with prominent neck dystonia and rigidity are characteristic of progressive supranuclear palsy. It is suggested that separate clinical evaluation of muscle tonus in the neck and trunk may be helpful for distinguishing progressive supranuclear palsy from Parkinson's disease.

Aged↗
PubMed1999

Identification of axial rigidity during locomotion in Parkinson disease.

OBJECTIVES: To identify coordination changes and stability in the movements of the trunk during locomotion in Parkinson disease (PD) as a function of walking velocity. STUDY DESIGN: Comparison of treadmill locomotion with an opto-electronic tracking device. PATIENTS: Newly diagnosed patients with PD (n = 27) and a group of healthy control subjects (n = 11). RESULTS: Coordination between transversal pelvic and thoracic rotations showed significantly smaller changes in mean relative phase (p < .0001) and lower variability in relative phase (p < .0001) in the PD group. No significant differences were found in stride duration and variability in stride duration. CONCLUSIONS: The relative phase data contradict traditional notions of increased variability in motor control in PD and pinpoint the importance of the trunk in identifying axial rigidity. This discrepancy may be due to lack of control for walking velocity in earlier studies. It is concluded that systematic manipulation of walking velocity can identify coordination deficits and rigidity in trunk movement. This coordination of trunk movement can also be a sensitive measure for (early) diagnosis and the assessment of movement and pharmacological therapy in PD.

Acceleration↗
PubMed2002

Biasing effect of the electromyogram on BIS: a controlled study during high-dose fentanyl induction.

OBJECTIVE: A biasing effect of the electromyogram (EMG) on the Bispectral Index (BIS) may explain discrepancies in previous studies assessing BIS in the presence of neuromuscular activity. Our aims were: to evaluate variations of BIS in the presence of high EMG activity associated with muscular rigidity after administration of high-dose fentanyl; to compare muscular rigidity, as measured by the EMG variable of the BIS monitor, in patients who were administered two different dosages of fentanyl at induction of cardiac anaesthesia. METHODS: 26 patients undergoing CABG surgery, after premedication with morphine 0.15 mg/kg, were randomized to receive either fentanyl 50 mcg/kg (group F) or fentanyl 10 mcg/kg plus etomidate 0.2 mg/kg (group EF). The induction dose was administered over 2 minutes. Patients were manually ventilated with O2 via face mask. Five minutes after induction was complete, patients were clinically assessed using the Responsiveness portion of the Observer's Assessment of Alertness/Sedation scale (OAAS). Haemodynamic data were recorded and arterial blood samples obtained at the time of OAAS observation. Patients were administered a neuromuscular blocking agent only after the OAAS assessment. BIS (3.4) was recorded from an A-2000 EEG monitor (Aspect Medical Systems) using disposable sensors (BIS Sensor, Aspect Medical Systems) applied per manufacturer's instructions. Data were recorded on a PC for off-line analysis. RESULTS: At the time of OAAS observation, mean (95% CI) BIS in group F was 85 (77-92) compared to 67 (56-79) in group EF (p = 0.01). Similarly, mean (95% CI) EMG was 50 dB (45-56) in F and 41 dB (35-47) in EF (p = 0.01). Correlation between BIS and EMG was very high (r2 = 0.88). OAAS scores were significantly higher in group F (p = 0.03). Non significant correlation was observed between BIS and OAAS scores (r2 = 0.32, p = 0.1). Backward stepwise multiple regression analysis including EMG, pH, CO2, O2 and OASS scores showed EMG as strong predictor of BIS (p < 0.0001, r2 = 0.7). Regression of EMG against BIS yielded the equation: BIS = 3.7 + (1.6 x EMG). CONCLUSION: During fentanyl-induced muscular rigidity BIS recordings reflect EMG variations. When assessing BIS in the absence of neuromuscular blockade, it is necessary to evaluate the effect of EMG on BIS before making conclusions about depth of sedation. Fentanyl-induced rigidity appears to be a dose-related phenomenon which the EMG variable of BIS 3.4 is able to quantify.

Aged↗
PubMed2003

Dopamine transporter binding in Wilson's disease.

INTRODUCTION: In Wilson's disease (WD), brain magnetic resonance images (MRI) show increased signal intensity in T2 weighted images in the lenticular nuclei, thalamus and brainstem, including the substantia nigra. A poor therapeutic response to levodopa in WD suggests the mechanism of a postsynaptic abnormality. However positron emission tomography studies show an involvement of the nigrostriatal presynaptic dopaminergic pathway. CASE REPORT: We report the clinical manifestations in a case of WD with akinetic-rigid syndrome and initial hesitation. The brain MRI showed an increased signal intensity lesion in the substantia nigra region, in addition to basal ganglion and thalamic lesions. However, dopamine transporter (DAT) imaging with 99mTc-TRODAT-1 revealed a nonsignificantly increased DAT uptake, suggesting a normal presynaptic nigrostriatal dopaminergic terminal. CONCLUSION: We suggest that significant heterogeneity can be found in WD patients and a normal presynaptic dopaminergic pathway may occur in some patients, even those with typical akinetic-rigid syndrome and evidence of substantia nigra involvement in the brain on MRI.

Adult↗