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PubMed1986

[A case of polyradiculoneuropathy associated with diffuse arterial narrowing].

A 32-year-old female was admitted complaining of pain over the whole body and dysesthesia in all extremities. On admission, physical examination revealed hypertension, tachycardia, abdominal distension and urinary retention. Neurological examination revealed sensory impairment over the whole body and mild muscular weakness with absent deep tendon reflexes. Abdominal roentgenogram showed gaseous distension of the bowels. CT scans showed low density areas on the bilateral parieto-occipital lobes. CAG showed diffuse narrowing of the cerebral arteries. Laboratory examinations showed an albumino-cytologic dissociation (cells; 21/3mm, prot.; 78 mg/dl) of the cerebrospinal fluid and increased levels of norepinephrine (214 micrograms/day) and epinephrine (16.1 micrograms/day) in the urine. Motor nerve conduction velocities of the median nerves was 42.8 m/sec in the right and 50.0 m/sec in the left. The autonomic function tests revealed hyper-responsiveness to 10 mg of phentolamine and low baroreflex sensitivity. Within 2 months, most symptoms improved. Repeated CT scans showed no abnormality, while CAG showed only mild arterial narrowing. This case was diagnosed as having acute autonomic and sensory neuropathy which was a subtype of acute polyradiculoneuritis. Occult encephalitis or autonomic dysfunction was considered to contribute to the peculiar findings on CT and CAG.

Adult↗

PubMed1993

Incapacitating autonomic neuropathy precipitated by taxol.

Taxol is one of the more significant advances in cancer chemotherapy in the last 10-15 years with a 30-50% response rate in both breast and ovarian cancers. The toxicity profile of taxol is still evolving as changes in the dosing and scheduling of the drug are instituted. We herein present the first evidence of taxol-induced quantifiable, objective autonomic neuropathy in two patients, one with and one without diabetes. The patients did not have symptoms of autonomic dysfunction prior to taxol therapy. Both developed severe orthostatic hypotension following completion of 24-hr infusional taxol at 170-250 mg/m2 and both improved markedly on fludrocortisone. Other possible etiologies of orthostatic hypotension were ruled out. Noninvasive autonomic tests performed on the first patient included tilt test, Valsalva ratio, heart rate variation with respiration, and change in diastolic blood pressure with hand grip; all were markedly abnormal. Noninvasive autonomic tests should be considered as part of the evaluation of patients on taxol who develop generalized weakness and orthostasis.

Aged↗

PubMed1996

3,4-Diaminopyridine, an orphan drug, in the symptomatic treatment of Lambert-Eaton myasthenic syndrome.

The Lambert-Eaton myasthenic syndrome (LEMS) is a rare autoimmune disease of peripheral cholinergic transmission that results in muscle weakness and autonomic dysfunction, due to impaired acetylcholine release. A review of available clinical information indicates that 3,4-diaminopyridine (3,4-DAP) used either alone or in conjunction with other therapies was effective in treating the motor and the autonomic deficits in patients with primary and paraneoplastic LEMS of varying degrees of severity. A survey of the medical literature indicates that about 150 patients have been treated worldwide with 3,4-DAP. The general view is that 3,4-DAP is well tolerated in short- or long-term treatments, with only mild side effects. 3,4-DAP is an orphan drug approved for clinical use in many european countries that lacks adoptive parents because its exploitation is not profitable.

4-Aminopyridine↗

PubMed1987

Synchronization of cardiac autonomic neural discharge with epileptogenic activity: the lockstep phenomenon.

Autonomic dysfunction has been implicated in the sudden, unexplained deaths which account for 5-17% of mortality in persons with epilepsy. This study was designed to determine if epileptogenic activity is associated with changes in the pattern of autonomic cardiac neural discharge and the development of arrhythmias. Nine cats, anesthetized with alpha-chloralose, received pentylenetetrazol (PTZ) 10, 20, 50, 100, 200 and 2000 mg/kg, i.v. at 10 min intervals. Cardiac postganglionic sympathetic and vagal nerve discharges were correlated with the interictal spikes, brief ictal discharges (bilateral polyspikes less than 10 sec duration), and prolonged ictal discharges (polyspikes lasting greater than 10 sec). Cardiac sympathetic and vagal neural discharges were intermittently synchronized 1:1 with all 3 types of epileptogenic discharge, i.e., the lockstep phenomenon (LSP); at other times the relationship was almost 1:1 LSP was not present during control and did not always persist for the entire interval after each PTZ dose. Five of 8 cats showed LSP in the cardiac sympathetic neural discharge associated with interictal spikes induced by 10 mg/kg PTZ; 3 others exhibited LSP with interictal spikes seen subsequent to ictal discharges. The incidence of LSP was less often associated with cardiac vagal neural discharge (2 of 7 cats). Premature ventricular contractions were sometimes associated with LSP. Abnormal cardiac sympathetic and vagal neural discharge and cardiac arrhythmias were thus associated with subconvulsant (interictal) activity. Therefore, the LSP may be a factor in the mechanism of unexplained death in persons with epilepsy who exhibited no overt seizure activity at the time of demise.

Animals↗

PubMed2004

Progressive ataxia and palatal tremor (PAPT): clinical and MRI assessment with review of palatal tremors.

Palatal tremor has been subdivided into essential (EPT) and symptomatic palatal tremor (SPT). A subgroup of the SPT form has a syndrome of progressive ataxia and palatal tremor (PAPT). Published details of cases of PAPT are sparse and the disorder appears heterogeneous. We present clinical and MRI features of six patients with sporadic PAPT who attended The University Health Network between 1991 and 2002. Eye movements were recorded using a magnetic search coil technique. We review previously reported cases of PAPT from the English language literature and relate this disorder to EPT and SPT. PAPT may be divided into sporadic and familial forms. We identified 22 other prior reported cases of sporadic PAPT. Sporadic PAPT is a subtype of SPT in which progressive cerebellar degeneration is the most symptomatic feature. A combination of vertical nystagmus and palatal tremor was found in one of our cases. Internuclear ophthalmoplegia, a new finding, was present in two of our patients and indicated additional brainstem dysfunction. Inferior olivary high signal abnormalities were present on MRI in all of our cases. The cause of sporadic PAPT remains uncertain. In some previous reports of sporadic PAPT, the combination of brainstem or pontine atrophy, parkinsonism, autonomic dysfunction or corticospinal tract abnormalities suggests a diagnosis of multiple system atrophy, although pathological verification is lacking. Familial PAPT is associated with marked brainstem and cervical cord atrophy with corticospinal tract findings, but the typical olivary MRI abnormalities have not been reported. A substitution in the glial fibrillary acidic protein (GFAP) gene has been described in a family with PAPT, raising the possibility of Alexander's disease. One other familial syndrome of PAPT, termed 'dark dentate disease', has also been reported. PAPT is a subgroup of SPT in which ataxia progresses and is not usually the result of a monophasic illness. Eye movement abnormalities suggest a disorder of both the cerebellum and brainstem. Familial PAPT differs from sporadic PAPT in having marked atrophy of cervical cord and brainstem with corticospinal signs but without hypertrophic olivary appearance on MRI.

Adolescent↗

PubMed2005

Comparison of postnatal development of heart rate responses to trigeminal stimulation in sleeping preterm and term infants.

Autonomic dysfunction has been regarded as a possible cause of the sudden infant death syndrome (SIDS) and it has been suggested that preterm infants, who are at a greater risk of SIDS than term infants, may have immature autonomic control. Our aim was to compare the maturation of cardiac autonomic control during sleep in preterm and term infants by examining heart rate responses to arousing and non-arousing trigeminal stimuli. Preterm infants (n = 15) and term infants (n = 24) were studied longitudinally with daytime polysomnography. Air-jet stimulation of the nares was delivered in both active sleep (AS) and quiet sleep (QS), and heart rate (HR) changes recorded for both arousal and non-arousal responses. Changes in HR (DeltaHR%) were calculated as the relative differences between baseline HR (BHR) and either MaxHR (arousal) or MinHR (non-arousal). Comparisons of HR changes between sleep states and postnatal ages were made with two-way anova for repeated measures and between groups with two-way anova. The increase in HR (DeltaHR%) was greater in term than preterm infants (P < 0.05), but only at 2-3 weeks corrected postnatal age (CPA). In preterm infants, there were no differences in BHR between sleep states, whereas in term infants, BHR was higher in AS than in QS at 2-3 weeks and 2-3 months of age. The smaller DeltaHR% to arousing stimuli in preterm infants compared with term infants at 2-3 weeks suggests that cardiac sympathetic activity in preterm infants may be lower than in term infants. This mechanism may account for the increased risk for SIDS of preterm infants.

Age Factors↗

PubMed1982

Reduced sinus arrhythmia in diabetic autonomic neuropathy: diagnostic value of an age-related normal range.

A study was carried out to establish a normal range for use in a deep breathing test of cardiac vagal integrity in diabetes mellitus. The change in heart rate resulting from taking a deep breath was recorded in 174 healthy subjects aged 16-89 years. Results were expressed as the ratio of the longest R-R interval during expiration to the shortest R-R interval during inspiration. This ratio declined appreciably with age (p less than 0.001) but was not significantly related to resting heart rate. An age-related normal range was constructed and its diagnostic value investigated in 134 diabetics (aged 15-70 years) with various degrees of neuropathy. The sensitivity for indicating autonomic dysfunction was high: six false-negative and one false-positive result were obtained. Measurement of this age-dependent ratio, which may be made with any electrocardiographic apparatus, provides a simple, accurate diagnostic screen for autonomic neuropathy in the clinic.

Adolescent↗

PubMed1996

[Familial amyloidotic polyneuropathy with a transthyretin variant (Val30-->Leu)].

We report a Japanese woman with familial amyloidotic polyneuropathy (FAP) with a transthyretin variant that substituted leucine for valine at position 30. Family history was not informative. She had initially suffered from repeated petechiae in the eyelids at the age of 51. Two years later, dysesthesia in the lower extremities appeared. Distal muscle weakness and sensory disturbance gradually developed. Autonomic dysfunction emerged and vomiting and orthostatic hypotension were marked in the late stage of her illness. Because of renal failure, she died at the age of 54. At autopsy, amyloid deposits were prominent in peripheral nerves including autonomic nerves, heart, blood vessels, gastrointestinal tract, kidneys, and bladder. In the nervous system, we found amyloid deposits in anterior roots, posterior roots, posterior root ganglions, peripheral nerves, and sympathetic ganglions. Cardiac weight was increased (595 g) with conspicuous amyloid deposits in the myocardium. The kidneys showed massive deposition of amyloid in the glomeruli and vascular walls. Amyloid accumulated moderately in tongue, submandibular gland, gallbladder, spleen, and pancreas, and slightly in the thyroid gland, lung, liver, and adrenal gland. No amyloid deposits were seen in the CNS with the exception of perivascular deposits in the choroid plexus.

Amyloid↗

PubMed2007

Autonomic function in sleep apnea patients: increased heart rate variability except during REM sleep in obese patients.

The objective of this study was to examine heart rate variability (HRV) among sleep stages in obstructive sleep apnea (OSA) patients. The study was retrospective within subjects and examined the sleep stages and HRV in relation to OSA, age, body mass index (BMI), and sex. Data collected during diagnostic polysomnograms were used in this study. There were 105 clinical patients undergoing polysomnography for suspected OSA. We sampled the electrocardiogram (ECG) from wakefulness, stage 2, and REM sleep and analyzed for frequency domain HRV. Sampled epochs were free of apnea and arousals. Heart rate variability decreased with age. Total frequency variability (TF) and low frequency variability (LF) in wakefulness and REM sleep increased as apnea severity increased. Measures of TF, LF, and the LF/HF ratio were greatest in REM sleep. There was less LF and TF in Stage REM sleep in patients with higher BMI. In conclusion, the decrease in HRV with aging is a robust finding that occurs even in a clinical sleep apnea population. However, apnea does not mimic aging effects on the heart because HRV increased as apnea severity increased. The decrease in HRV during REM sleep in the obese apnea patients suggests the possibility of an autonomic dysfunction in this subgroup.

Adult↗

PubMed1989

Autonomic and myocardial changes in middle cerebral artery occlusion: stroke models in the rat.

Stroke models in larger animals such as the cat, dog and monkey are becoming increasingly more expensive and less readily available. However, the rat is an excellent model for focal cerebral ischemia. Rats are readily available, inexpensive and their neuroanatomy and brain function have been studied extensively. Increases in plasma catecholamines and myocardial damage have been observed in clinical stroke. We examined autonomic and myocardial changes in two rat stroke models. In one model only the middle cerebral artery was occluded (MCAO) while the other model involved occlusion of both the MCA and the common carotid artery (MCAO/CCAO). Arterial blood pressure and heart rate were monitored continuously in 25 male rats (326-430 g) that underwent one of the following procedures: (1) MCAO only; (2) MCAO/CCAO; (3) CCAO only; and (4) sham occlusions (SHAM). Arterial blood samples (0.5 ml) for radioenzymatic assay of norepinephrine (NE) and epinephrine (E) were taken twice before the occlusions and at 90 and 180 min after the occlusions. The animals were perfused at the end of the experiment and the heart removed and examined histologically. Tetrazolium salts were reacted with oxidative enzymes to delineate the region of inadequate perfusion. The mean blood pressure and pulse pressure of the SHAM, MCAO/CCAO and CCAO groups significantly declined from initial values (from an average of 78 to 53 mm Hg) during the course of the experiment. However, the mean blood pressure and pulse pressure of the MCAO rats did not change during the experiment, so that the final mean blood pressure and pulse pressure were significantly higher than in the other 3 groups. The levels of both NE and E increased significantly (NE, 1443 +/- 285.9 to 4095 +/- 929 pg/ml; E, 2402 +/- 623 to 3741 +/- 1166 pg/ml) following occlusion in the MCAO group only while the other 3 groups did not change. Four of 6 hearts in the MCAO group were abnormal, showing evidence of subendocardial hemorrhage, ischemic damage or subendocardial congestion. MCAO also resulted in a consistent region of the brain with inadequate perfusion including the insular cortex. These autonomic and myocardial changes appear to mimic some of the changes seen clinically in stroke patients and provide the first acute stroke model for studying autonomic dysfunction in the rat.

Animals↗

PubMed2004

Nonhypotensive autonomic agents in veterinary ophthalmology.

The parasympathetic and sympathetic divisions of the autonomic nervous system are involved in homeostatic control of a wide variety of ocular functions, including accommodation, pupillomotor control, lacrimation, eyelid position, and aqueous humor production. Familiarity with the functional anatomy of the autonomic nervous system is paramount to the understanding and application of the large number of autonomic drugs used in veterinary ophthalmology. The cholinergic and adrenergic agents discussed in this article are commonly employed to facilitate routine ophthalmic examination, in the diagnosis of autonomic dysfunction, and in the treatment of a variety of ocular diseases.

Administration, Topical↗

PubMed1988

High frequency of autonomic as well as peripheral neuropathy in patients with malnutrition-related diabetes mellitus.

We assessed peripheral and autonomic nerve function in 27 diabetics. Ten had malnutrition-related diabetes mellitus (MRDM), eight insulin-dependent diabetes mellitus (IDDM), and nine non-insulin-dependent diabetes mellitus (NIDDM). The frequency of peripheral neuropathy was 70, 78 and 13% in MRDM, NIDDM and IDDM respectively. Furthermore, the frequency of abnormality in cardiac beat-to-beat variation was 50 and 38% in MRDM and IDDM respectively, whereas our NIDDM patients did not show this abnormality. The patients with MRDM thus revealed a high frequency of not only peripheral but also autonomic neuropathy. Autonomic dysfunction appears to be a new characteristic of MRDM, not mentioned by the WHO study group.

Autonomic Nervous System↗

PubMed1992

Dural tear following traumatic spinal cord injury. A delayed complication.

Dural tears with leakage of cerebral spinal fluid into surrounding soft tissues can occur after traumatic spinal cord injury. An unusual case presented in a patient with traumatic paraplegia where the onset was delayed and clinical features were suggestive of autonomic dysfunction. The clinical features, pathophysiology and treatment of this interesting complication following traumatic spinal cord injury are discussed.

Adult↗

PubMed1991

The influence of food on postural hypotension in three groups with chronic autonomic failure--clinical and therapeutic implications.

The effect of a balanced liquid meal on supine and postural blood pressure (BP) responses was investigated in three groups of patients with chronic autonomic failure; 10 with associated neurological impairment (multiple system atrophy (MSA), Shy-Drager syndrome) and seven without (of which five had pure autonomic failure (PAF); and two had a deficiency of the enzyme dopamine beta hydroxylase, DBH-deficiency). All had marked postural hypotension. Subjects with normal autonomic function were also studied. In MSA and PAF food lowered supine BP substantially, with a more rapid and greater fall in PAF. After food, the levels of BP reached were considerably lower because of the reduced supine BP and many had to be returned to the horizontal position earlier than before. Ingestion of a similar volume of water alone had no effect in MSA or PAF. In DBH deficiency, food had variable but minimal effects on BP while supine and during head-up tilt. In subjects with normal autonomic function food did not affect BP. The BP responses to food thus varied in the three groups with chronic autonomic failure. The influence of food on both supine and postural BP therefore should be considered in the clinical and laboratory assessment of autonomic dysfunction and in relation to therapeutic approaches, designed to alleviate postural hypotension.

Adult↗

PubMed2001

[Hematosalivary mechanisms of regulation in rheumatoid arthritis].

AIM: To study biochemical indicators of the hematosalivary barrier operation in varying activity of articular inflammation and their correlations. MATERIAL AND METHODS: Biochemical indices of mixed saliva and peripheral blood were studied in 38 healthy subjects and 99 patients with rheumatoid arthritis (RA). RESULTS: Optimal composition of the saliva was abnormal. It lost its buffer function. In RA, permeability of the hematosalivary barrier for cholesterol, calcium, phosphorus, magnesium declines with growing activity of the process with resultant lowering of their content in the saliva, their blood levels being unchanged. CONCLUSION: Decreased permeability of the hematosalivary barrier results from growing sympathetic influence on the salivary glands reflecting the presence of the autonomic dysfunction in RA. It is pathogenetically related to the process activity.

Adult↗

PubMed2005

Brain natriuretic peptide and cardiac autonomic function in type 2 diabetic patients.

The present study tested the hypothesis that increased plasma brain natriuretic peptide (BNP) levels are related to cardiac autonomic dysfunction in type 2 diabetic patients. A total of 32 consecutive Japanese patients with type 2 diabetes were assigned to either a high-BNP (>or=18 pg/ml) group (n=12; age 57+/-13 years, mean+/-S.D.) or a normal-BNP (<18 pg/ml) group (n=20; 59+/-10 years). No patient had any overt structural heart disease. Cardiac autonomic function was assessed by measurements of baroreflex sensitivity (BRS), heart rate variability (HRV) and cardiac (123)I-metaiodobenzylguanidine (MIBG) scintigraphic findings. BRS was lower (p<0.005) in the high-BNP group than in the normal-BNP group. However, the components of HRV, and the early and delayed myocardial uptake of (123)I-MIBG and percentage washout rate of (123)I-MIBG were not significantly different between the groups. The plasma level of BNP negatively correlated with BRS (r=0.35, p=0.049). These findings suggest that increased plasma BNP levels were related to cardiac reflex parasympathetic dysfunction in our Japanese type 2 diabetic patients.

3-Iodobenzylguanidine↗

PubMed2001

Autonomic and cardiovascular function in HIV spectrum disease: early indications of cardiac pathophysiology.

Autonomic dysfunction in persons with acquired immune deficiency syndrome (AIDS) has been reported previously but its incidence in early stage HIV infection and its relation to cardiovascular function have not been fully examined. The present study evaluated cardiovascular and autonomic function in 55 HIV-seronegative, and 52 HIV-asymptomatic and 31 HIV-symptomatic seropositive men. Measures of hemodynamic and autonomic function were obtained at rest and during a standardized battery of autonomic tests. Results were compared across groups while controlling for age, body mass, and physical activity. Analyses indicated that measures of autonomic function did not differ among groups. However, at rest, both HIV seropositive groups exhibited diminished stroke volume and elevated diastolic blood pressure, albeit within normotensive levels. In addition, the ability to sustain a blood pressure response during prolonged challenge and the relationship between stroke volume and baroreceptor/vagal responsiveness were disrupted in the HIV-symptomatic group. Therefore, in the pre-AIDS stages of infection, autonomic functioning appeared intact; yet alterations in baroreceptor/vagal function associated with depressed myocardial function may be an early warning signal reflecting cardiovascular pathological processes potentially exacerbated by HIV spectrum disease.

Adult↗

PubMed2006

Skin biopsy for assessment of autonomic denervation in Parkinson's disease.

Autonomic dysfunction in Parkinson's disease (PD) is considered a late complication of the disease or an adverse effect of anti-parkinsonian medications. Morphological changes are demonstrated only by postmortem examination. The study objective was to evaluate peripheral autonomic neural involvement in PD using punch skin biopsy. The study sample included 22 patients (mean age 50 +/- 7.7 years, mean disease duration 5.3 +/- 3.8 years) and 19 controls. Four-millimeter skin biopsies were immunohistochemically stained with anti-PGP 9.5 antibody. Autonomic innervation of the blood vessels, sweat glands, and erector pili muscles was assessed and rated from 0 (normal) to 2 (severe). Cutaneous autonomic innervation was decreased in patients compared to controls. Semi quantitative analysis demonstrated reduced autonomic innervation of the blood vessels (1.0 +/- 0.8 vs. 0.42 +/- 0.8 in controls; p < 0.02), of sweat glands (0.95 +/- 0.67 vs. 0.47 +/- 0.61; p < 0.02) and of the erector pili muscles (1.06 +/- 0.55 vs 0.21 +/- 0.42; p < 0.001). This method demonstrates that the peripheral autonomic system is affected in PD at early stage of the disease and that autonomic involvement in PD may be more prevalent than previously thought.

Adult↗