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PubMed2004

Gliomatosis cerebri may present as an atypical parkinsonian syndrome.

An 85-year-old man presented with 3 years of a progressive akinetic rigid syndrome with ataxia, falls, levodopa unresponsiveness, and autonomic dysfunction. Early in the course of illness, cranial magnetic resonance imaging (MRI) was thought to show pontine enlargement but later was reinterpreted by experts as normal. Two subsequent cranial MRIs were believed to show only mild atrophy. An initial clinical diagnosis of multiple system atrophy was made. The patient died 4 years after the onset of symptoms. Pathological examination revealed diffuse infiltration of multiple brain regions with cells of astrocytic and microglial origin consistent with a diagnosis of gliomatosis cerebri.

Aged↗

PubMed1984

Sinus arrest during REM sleep in young adults.

Four apparently healthy young adults with vague chest symptoms during the day, two of whom had infrequent syncope while ambulatory at night, had periods of asystole up to nine seconds in duration occurring repeatedly during rapid-eye-movement (REM) sleep. Extensive evaluations, including electrophysiologic studies in two patients, were normal. It is therefore suggested that the underlying pathophysiology involved autonomic dysfunction. REM sleep-related sinus arrests such as these, which may occur in apparently healthy subjects but are undetected, may explain some cases of sudden, unexpected death during sleep. Polygraphic monitoring during sleep may be helpful in delineating the pathophysiology of the sleep-related arrhythmia in persons with daytime cardiac arrhythmias thought to be secondary to abnormal vagal tone. The possibility of nocturnal asystole should be considered in patients such as those described here.

Adult↗

PubMed1988

Serum antibodies to brain proteins in a patient with parkinsonism associated with IgM paraproteinemia.

We report a 58 year-old man with various neurological symptoms as parkinsonism, autonomic dysfunction, mental deterioration and lower motor neuron involvement associated with IgM gammopathy whose serum showed antibody activities to human brain proteins. His serum reacted with 156kDa protein from substantia nigra and 130kDa of cerebral cortical grey matter, cerebellar cortex, putamen, thalamus or pallidum by immunoblotting method. The serum of this patient reacted with proteins widely distributed in the central nervous system, and this antibody in his serum might be the cause of his various neurological symptoms.

Autoantibodies↗

PubMed1999

Neonatal Hirschsprung disease, dysautonomia, and central hypoventilation.

BACKGROUND: A reported association between congenital central hypoventilation, long-segment intestinal aganglionosis (Hirschsprung disease), and autonomic dysfunction, with a high recurrence risk and mortality rate, is associated with abnormal neural crest development (neurocristopathy). CASE: A fetus had increasing polyhydramnios, no stomach bubble, and repeatedly nonreactive fetal heart rate tracings despite normal activity. There were no other fetal anomalies on ultrasound. Postnatally all of the above clinical features were diagnosed, prompting diagnosis of neurocristopathy syndrome. She died at 2 weeks of age. CONCLUSION: Antenatal polyhydramnios, nonreactive nonstress tests, and absent stomach bubble in an active fetus indicated neurocristopathy.

Adult↗

PubMed2003

Gastric emptying time and the effect of cisapride in cirrhotic patients with autonomic neuropathy.

GOALS To investigate the relationships between gastric emptying and autonomic dysfunction in hepatic cirrhosis and to assess the effects of cisapride on gastric emptying in cirrhotic patients. STUDY Twenty-four cirrhotic patients (8 patients in each Child-Pugh classification) and 25 healthy controls were enrolled. All the patients had viral (B or C) hepatitis. Patients with DM, alcoholic cirrhosis, active peptic ulcer, gastric malignancy and pyloric obstruction were excluded by esophagogastroduodenoscopy. Parasympathetic and sympathetic functions were assessed by the criteria set forth by Ewing and Clark. Drugs affecting GI motility and smoking were discontinued 48 hours and 12 hours prior to the study respectively. A solid-phase of gastric emptying study was conducted by scintigraphic method for the calculation of gastric half-emptying time (GET1/2). RESULTS The study revealed that 9 patients with Child-Pugh B and C cirrhosis had autonomic neuropathy and none of Child-Pugh A cirrhosis had autonomic neuropathy. Prolonged GET1/2 was noted in cirrhotics compared with the control group (p < 0.05). However, there was significant difference between 9 patients (Child B-C) with autonomic neuropathy compared with patients 15 patients without autonomic neuropathy. Again there was a significant difference in GET1/2 between Child A cirrhotic and Child B-C cirrhotic whether they had autonomic neuropathy or not. Cisapride decreased GET1/2 significantly in cirrhotic patients (Child B-C cirrhotic). Clearly, patients with autonomic neuropathy in Child B-C cirrhosis had significantly reduced GET1/2 after cisapride administration. Even though cisapride decreased GET1/2 in patients with Child B-C cirrhosis without autonomic neuropathy, this was not significant. CONCLUSION Autonomic neuropathy in advanced cirrhosis from viral hepatitis may cause prolonged gastric emptying. Cisapride can shorten gastric emptying time in such patients.

Administration, Oral↗

PubMed1994

Exercise and immunology.

The documented effects of physical activity on host defense date to the early 1900s, although the influences of exercise on infectious and neoplastic disease susceptibility in healthy persons have long been subject to widespread disagreement. Even less is known of the effects of exercise in reducing illness predilection for persons already susceptible to, or afflicted by, immune disease or dysfunction. Recently, however, reports suggest that exercise may reduce the incidence and severity of infection. These reports have been bolstered by well-conducted epidemiologic and laboratory studies demonstrating a link between moderate exercise and either heightened immune function or reduced disease incidence, a relationship that is reversed in instances of immoderate exercise. This symposium will present current information addressing beneficial and detrimental influences of exercise on immune function and disease susceptibility. The authors will emphasize direct effects of acute exercise and physical training on immune cells and their neuroendocrine/immune modulators. The relationship among exercise, infection susceptibility, and immune system function will be highlighted, as well as exercise-induced activation of, and illness/disease regulation by, natural killer cells. Consequences of exercise on immune function and disease advancement will be addressed for persons with neoplasia, autonomic dysfunction, aging-related immunosenescence, and AIDS.

Disease Susceptibility↗

PubMed2002

Heart rate response profiles to tilting in healthy and unhealthy neonates.

BACKGROUND: Clinical observations suggest the immaturity of the neonate's ability to respond appropriately to cardio-respiratory challenges and tilt test could help in detecting autonomic dysfunction in neonates. MATERIAL/METHODS: Heart rate (HR) responses following a 45 degrees head-up tilt during sleep were measured within the first week of adjusted age in 7 healthy full-term neonates, 7 healthy preterm infants and 5 neonates with abstinence syndrome. RESULTS: The tilt and the return to the horizontal position generally provoked a sustained or transient tachycardia, or a flat response in healthy full-term neonates and in preterm neonates, but a sustained or transient bradycardia in neonates with abstinence syndrome. CONCLUSIONS: When compared to previous studies in older infants, our data point to the immaturity of the neonate's ability to respond appropriately to cardio-respiratory challenges. No difference is noted between full-term and preterm neonates. The altered HR response in neonates with abstinence syndrome suggests an autonomic dysregulation in this group.

Bradycardia↗

PubMed1986

[Dysautonomia at 2 times in Guillain-Barré syndrome].

Autonomic dysfunction is described in a 63 year old woman suffering from Guillain-Barré syndrome. Bradyarrhythmias occurred during the convalescent phase, whereas neurologic and respiratory symptoms were improving; they were not related to bronchial aspiration. Their severity and the inefficiency of medical treatment led to the insertion of a demand pace-maker. Cardiac monitoring by the Holter method in severe cases of Guillain-Barré syndrome may detect potentially lethal arrhythmias, that may then be avoided by a demand pace-maker.

Autonomic Nervous System Diseases↗

PubMed1992

Diurnal blood pressure variability in mineralocorticoid excess syndrome.

Noninvasive 24-h blood pressure (BP) monitoring has demonstrated a diurnal blood pressure profile in most individuals that is characterized by higher arterial pressures during wakefulness and lower pressures at night during sleep. Recently, reports suggest that this typical diurnal variation is absent in syndromes of autonomic dysfunction and in some forms of secondary hypertension. We investigated the 24-h BP, BP variability, and adrenal steroid concentrations in a patient with deoxycorticosterone (DOC)-secreting adrenal adenoma prior to and following adrenalectomy. Preoperatively, when the patient had a ten-fold increase in serum concentrations of DOC, there was no fall in nocturnal BP despite a marked reduction in heart rate during sleep. Postoperatively, when the concentrations of DOC and other adrenal steroids returned to normal values, the 24-h BP profile normalized with restoration of the nocturnal reduction in pressure. These findings document the effects of mineralocorticoid overproduction on diurnal BP regulation. Intensive investigation of individuals with a well-defined etiology of hypertension and the absence of diurnal variation of BP may lead to further hypotheses that will define the role of both autonomic and nonautonomic factors in BP control.

Adenoma↗

PubMed1988

Dysautonomia in a cat.

Autonomic dysfunction was diagnosed in a 2.5-year-old spayed domestic shorthair cat. The cat had an 8-day history of progressive anorexia, signs of depression, constipation, weight loss, and intermittent regurgitation. Physical examination findings were signs of depression, dehydration, cachexia, bradycardia, bilateral nonresponsive mydriasis, prolapse of both nictitating membranes, dry oral and nasal mucous membranes, and urinary bladder atony. Thoracic radiography revealed megaesophagus. The cat lacked esophageal motility and had a decreased gastric emptying rate. Providing adequate fluid intake, electrolyte balance, and nutrition is a major problem in the management of dysautonomic cats. We were able to provide adequate nutritional support for this patient, using total parenteral feeding and, later, enteral nutrition using a nasogastric tube. Results of an ocular pharmacologic study indicated that the mydriasis and prolapse of the nictitating membrane were attributable to complete autonomic denervation of the eye. Using the method described, topical, autonomic-stimulating agents may assist the clinician in diagnosing dysautonomia in the feline. This report describes a syndrome that is well recognized in the United Kingdom and has the potential to develop in the United States.

Animals↗

PubMed2004

Prognostic value of heart rate variability after acute myocardial infarction.

BACKGROUND: Prognosis after acute myocardial infarction (AMI) may be influenced by autonomic dysfunction that can be evaluated by assessment of heart rate variability (HRV). Its predictive value resulted from studies performed prior to large scale use of reperfusion therapy. We assessed the prognostic value of HRV parameters 1 year after AMI in patients treated conventionally or by a reperfusion method in the first 12 hours from onset. MATERIAL/METHODS: We included 463 consecutive patients with AMI (312 M, 151 F) 60.6+/-13.0 years old. 211 were treated by thrombolysis or primary PTCA, the other 251 patients receiving conventional therapy. Time-domain (SDNN, rMSSD) and frequency-domain (LF, HF, total power) HRV parameters were calculated from 24-hour Holter ECG recordings 10-20 days after AMI. The primary endpoint was one-year total mortality and sudden cardiac death. RESULTS: The incidence of cardiac death was 14.7%, while that of sudden death was 4.8%. Both were higher in patients treated conventionally. Patients treated by reperfusion had higher HRV parameters reflecting both vagal and sympathetic activity (SDNN, total spectral power) as well as those expressing only vagal output (rMSSD, HF power) than conventionally treated subjects. The variables independently correlating with 1-year survival were SDNN<50 msec, rMSSD<20 msec, LF/HF>2, non-sustained ventricular tachycardia, and left ventricular ejection fraction <40%. CONCLUSIONS: HRV parameters have prognostic value independent from left ventricular ejection fraction and spontaneous ventricular arrhythmias one year after AMI. Reduction of mortality risk by reperfusion therapy does not decrease the prognostic utility of HRV after AMI.

Aged↗

PubMed1991

Spectral analysis of heart rate in diabetic autonomic neuropathy. A comparison with standard tests of autonomic function.

Power spectral analysis of the heart rate was compared with standard tests of autonomic function performed on 15 insulin-dependent diabetic patients with symptoms of autonomic dysfunction. The high-frequency power, low-frequency power, and total power of the heart rate power spectrum measured in the supine position--possible measures of parasympathetic nervous system function--were significant predictors of the expiratory-inspiratory ratio, the beat-to-beat heart rate difference on deep respiration, the standard deviation of the resting heart rate, the 30:15 ratio, and the Valsalva ratio. The change in low-frequency power on moving from the supine to upright position, a possible measure of sympathetic nervous system function, was a modest predictor of the systolic and diastolic blood pressure fall in response to postural change, but a poor predictor of the blood pressure response to isometric exercise and a cold stimulus. This study supports the role of heart rate power spectral analysis as a clinical test of autonomic function in patients with known or suspected autonomic failure.

Adult↗

PubMed1999

[Severe forms of eosinophilic meningitis in infants of Mayotte. Apropos of 3 cases].

BACKGROUND: Eosinophilic meningitis caused by Angiostrongylus cantonensis is widespread in Southeast Asia and the Pacific islands. Adults develop transient meningitis with a benign course, whilst severe or fatal disease may occur in pediatric patients. CASE REPORTS: Three infant girls, aged 8 to 11 months, living on the island of Mayotte, developed fever, hypotonia, coma (2 cases), and, for one of them, seizures. Eosinophilia was detected in the peripheral blood and cerebrospinal fluid. Secondary, flaccid quadraplegia (1 case) or paraplegia (2 cases) with absence of deep tendon reflexes, urinary retention and anal incontinence were noted. Three patients had autonomic dysfunction. Computerized tomography showed enlarged ventricles and cerebral subarachnoid spaces. One patient had sequelae. Two patients could not be followed. Retrospectively, the diagnosis of angiostrongylus infection was established for two infants by a serological study. CONCLUSION: We report three new cases of infants with severe Angiostrongylus cantonensis infection in the French island of Mayotte (Comoro Islands). In this Indian Ocean area, eosinophilic meningitis seems to occur exclusively in infants and with severe radiculomyeloencephalitic forms.

Angiostrongylus cantonensis↗

PubMed1999

Decreased heart rate variability in patients with type 1 diabetes mellitus is related to arterial wall stiffness.

OBJECTIVE: Low heart rate variability (HRV) is, in several patient groups, related to poor prognosis. The underlying mechanisms are still unclear. The aim was to study if there is a relationship between HRV, which is a measure of baroreceptor function, and atherosclerosis. DESIGN: The relationship between heart rate variability and carotid arterial wall stiffness was studied in subjects with type 1 diabetes mellitus in which autonomic dysfunction and early atherosclerosis are common. HRV was assessed from power spectral analysis of 24-h Holter recordings and arterial wall stiffness was assessed from an ultrasound study of the right common carotid artery. SETTING: A university hospital. SUBJECTS: Fifty-nine patients (41 +/- 8 years) from the Stockholm Diabetes Intervention Study (SDIS) were investigated. These patients were randomized to intensified conventional treatment or standard treatment approximately 12 years before this study. RESULTS: Patients with stiffer arteries had lower HRV in all spectral bands (r = -0.32 to -0.40, P = 0.06-0.001). This relation remained on correcting for age. All spectral parameters of HRV correlated with the mean HbA1c from 10 years of study (r = -0.37 to -0.40, P = 0.004-0.001). CONCLUSIONS: In patients with type 1 diabetes mellitus, heart rate variability and arterial wall stiffness are related to each other. The results suggests that the autonomic nervous system could be a link between diabetes and vascular disease.

Adult↗

PubMed1994

Immune responses to nervous system decentralization and exercise in quadriplegia.

Interactions among the nervous, neuroendocrine, and immune systems render host defenses highly sensitive to autonomic over- or understimulation. Persons with quadriplegia experience decentralization of directly innervated immune tissues and neuroendocrine axis dysregulation, immobilization deconditioning, heightened exposure to immune suppressing xerobiologicals, and psychic and nonpsychic stressors differing from those of nondisabled cohorts. When compared with matched nondisabled controls, young survivors of quadriplegia have reduced CD4:CD8 ratios, suppressed proliferative responses to mitogen challenge, reduced number and cytotoxicity of CD3-CD56+ (NK) cells, and elevation of the soluble IL-2 receptor. Deviations from control values are typically observed in persons with injuries higher than sympathetic outflow, suggesting a cause related to autonomic dysfunction. Cycling exercise performed by persons with quadriplegia using computer-sequenced electrically stimulated contraction of the quadriceps, hamstring, and gluteus muscle groups fails to provoke an archetypical leukocytosis, but transitionally elevates NK cell number and cytotoxicity lasting one-half hour after exercise. These findings show that the immune system of persons with quadriplegia is selectively responsive to exercise challenge. As opportunistic infections of the urinary tract, lungs, and skin are major causes of morbidity in survivors of quadriplegia, these observations may identify a treatment through which their host defenses can be fortified.

Autonomic Nervous System↗

PubMed1985

The use of magnesium sulphate infusions in the management of very severe tetanus.

The use of magnesium sulphate infusions as an adjunct to the management of patients with very severe tetanus was studied in 10 subjects with persistent autonomic dysfunction despite heavy sedation. Satisfactory cardiovascular control was established in nine of these cases, and there were two deaths in the series, neither directly attributable to the tetanus itself. Haemodynamic measurements performed in six patients, showed that cardiac output was well preserved during magnesium infusions. There was a significant reduction in pulse rate (p less than 0.05) and systemic vascular resistance (p less than 0.01), with an increase in stroke volume (p less than 0.05). It is suggested that magnesium may be a useful adjunct to standard forms of management of such cases.

Autonomic Nervous System Diseases↗

PubMed1989

[Orthostatically induced epileptoid attack].

An epileptoid attack induced by orthostatic hypotension seen in a 72-year-old man was reported. The patient had been suffering from progressive autonomic failure with parkinsonism for six years and he had severe orthostatic hypotension, syncope and generalized convulsion when he stood up. The convulsion sometimes associated with urinary incontinence, ceased immediately when he lay down although he remained drowsy for a while. Occasionally the seizure ceased spontaneously and he regained consciousness even while he was kept standing. On lying position his blood pressure was 167/88 mmHg and no abnormality was seen in electroencephalogram. When he was tilted up to 50 degrees his blood pressure fell to 70/46 mmHg, and he became unconscious followed by jaw twitching and generalized clonic seizure. Electroencephalogram during seizure showed sharp wave and rhythmic spikes. Other laboratory examination revealed diffuse and severe autonomic dysfunction and slight cerebral atrophy on brain CT scanning. He was treated with diphenylhydantoin 300 mg/day and the seizure responded partially. The reasons why the patient's seizure was thought to be epileptic rather than the convulsive syncope were as follows: the type of the seizure was similar to an epileptic generalized convulsion, the seizure and unconsciousness ceased spontaneously even during standing position, the seizure and impaired consciousness partially responded to diphenylhydantoin administration. The underlying pathophysiology of the seizure was thought to be transient cerebral hypoperfusion induced by orthostatic hypotension.

Aged↗

PubMed1996

Ventilatory response to exercise in diabetic subjects with autonomic neuropathy.

We have used diabetic autonomic neuropathy as a model of chronic pulmonary denervation to study the ventilatory response to incremental exercise in 20 diabetic subjects, 10 with (Dan+) and 10 without (Dan-) autonomic dysfunction, and in 10 normal control subjects. Although both Dan+ and Dan- subjects achieved lower O2 consumption and CO2 production (VCO2) than control subjects at peak of exercise, they attained similar values of either minute ventilation (VE) or adjusted ventilation (VE/maximal voluntary ventilation). The increment of respiratory rate with increasing adjusted ventilation was much higher in Dan+ than in Dan- and control subjects (P < 0.05). The slope of the linear VE/VCO2 relationship was 0.032 +/- 0.002, 0.027 +/- 0.001 (P < 0.05), and 0.025 +/- 0.001 (P < 0.001) ml/min in Dan+, Dan-, and control subjects, respectively. Both neuromuscular and ventilatory outputs in relation to increasing VCO2 were progressively higher in Dan+ than in Dan- and control subjects. At peak of exercise, end-tidal PCO2 was much lower in Dan+ (35.9 +/- 1.6 Torr) than in Dan- (42.1 +/- 1.7 Torr; P < 0.02) and control (42.1 +/- 0.9 Torr; P < 0.005) subjects. We conclude that pulmonary autonomic denervation affects ventilatory response to stressful exercise by excessively increasing respiratory rate and alveolar ventilation. Reduced neural inhibitory modulation from sympathetic pulmonary afferents and/or increased chemosensitivity may be responsible for the higher inspiratory output.

Adult↗