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PubMed1999

Cardiovascular autonomic function in patients with relapsing remitting multiple sclerosis: a new surrogate marker of disease evolution?

Twenty patients with active relapsing remitting multiple sclerosis (MS) were examined annually for 2 years with a set of autonomic function tests (AFT) consisting of heart rate variability during deep breathing (IE), standing-up, and ratios of Valsalva manoeuvre (VR). Disease characteristics, including T2-weighted magnetic resonance imaging (MRI) of the brain and the expanded disability status scale (EDSS) score were documented each year within 1 week of the AFT. The EDSS score, MRI load lesion and VR did not change significantly over the follow-up period. The IE and initial heart-rate on standing during the first 30 s (DeltaHRMAX) showed significant worsening during follow-up. No relationship was found between deterioration of AFT and EDSS score, number of exacerbations, duration of disease, gender, age, size and number of lesions on MRI. We conclude that patients with active relapsing remitting MS show progression of autonomic dysfunction over a relatively short time. Therefore, in the absence of changes in clinical disability or brain MRI lesion load, AFT might be useful as a sensitive surrogate outcome measure for demonstrating subclinical change in MS.

Adult↗

PubMed2001

[Acupuncture correction of autonomic nervous system disorders in patients with bronchial asthma].

Acupuncture was added to medicines in the treatment of 192 patients suffering from bronchial asthma with autonomic dysfunction. The effect was assessed by changes in rhythmogram, external respiration function, psychological status. It was found that corporal acupuncture not only improves bronchial permeability but also reduces psychovegetative disorders. The effect of acupuncture was not related to that of placebo.

Acupuncture Therapy↗

PubMed1981

Postsynaptic adrenergic unresponsiveness in hypotensive haemodialysis patients.

In order to ascertain the significance of autonomic dysfunction in hypotensive maintenance haemodialysis patients, plasma norepinephrine (NE) was measured by radioenzymatic assay in 15 nondiabetic uraemic subjects of similar age and time on dialysis, five of them chronically hypotensive and 10 normotensive. Hypotensive patients had: 1) Significantly higher NE levels than their normotensive counterparts, both in basal conditions and after orthostatism and 2) significantly lesser increments in mean arterial blood pressure during a NE infusion than the normotensive group. Rather than implicating a mechanism based on autonomic neuropathy, our results suggest that in haemodialysis patients with chronic hypotension, there is a defect located at the adrenergic postsynaptic level.

Adult↗

PubMed2003

Clinicopathologic and genetic features of early- and late-onset FAP type I (FAP ATTR Val30Met) in Japan.

Type I (transthyretin Val30Met) familial amyloid polyneuropathy (FAP ATTR Val30Met) has been reported in relation to two endemic foci in Japan. These cases are characterized by a relatively young age at onset, between the second and third decade, high penetrance rate, concentration in endemic foci, predominant loss of superficial sensation, severe autonomic dysfunction, and atrioventricular nodal block requiring pacemaker implantation. In contrast to these endemic cases, because of advances in DNA diagnosis, late-onset cases of FAP ATTR Val30Met with symptoms appearing at or over 50 years of age are now recognized to occur widely throughout Japan. These cases have a male preponderance, low penetrance rate, no relationship to endemic foci, sensorimotor symptoms beginning distally in the lower extremities with disturbance of both superficial and deep sensation, and relatively mild autonomic symptoms. This type of FAP ATTR Val30Met has been overlooked because its clinical and genetic features differ from those of "typical" early-onset cases. Anticipation of age at onset is known to occur in pedigrees from the two endemic foci in Japan, with age at onset becoming younger in patients of successive generations. On the other hand, age at onset of patients in late-onset families seems to be uniformly late among the patient siblings when family history is present. Pathologic findings of the peripheral nervous system also differ in accordance with differences of clinical features. Loss of dorsal root and sympathetic ganglion neurons was severe in the early-onset cases, whereas it was only mild to moderate in the late-onset cases. Unmyelinated fibers in the biopsied sural nerve specimens of late-onset cases seemed to be relatively well preserved compared to those of previously reported early-onset cases.

Age of Onset↗

PubMed2007

Clinical variability in type I familial amyloid polyneuropathy (Val30Met): comparison between late- and early-onset cases in Portugal.

We compared early- with late-onset cases in 86 patients with familial amyloidotic polyneuropathy type I (FAP). Among these patients, 43 presented before age 50 (early-onset) and 43 after this age (late-onset). Sex and geographical distribution were similar, although a family history of the disorder was more frequent in early-onset cases. In late-onset disease, autonomic dysfunction was less frequent, but organ involvement and severe neuropathic pain were more frequent. Neurophysiological measurements were similar. FAP is a cause of neuropathy in elderly patients, in particular in those presenting with neuropathic pain.

Adult↗

PubMed1998

Vecuronium dose requirement and pupillary response in a patient with olivopontocerebellar atrophy (OPCA).

PURPOSE: Olivopontocerebellar atrophy (OPCA), a variant of spinocerebellar degeneration (Shy-Drager syndrome), is a systemic degenerative disorder affecting the neurons of multiple nuclei. We investigated the sensitivity to vecuronium and the pupillary responses to various stresses in a patient with OPCA. CLINICAL FEATURES: A 65-yr-old woman with a six-month history of OPCA underwent a left upper lobectomy for lung cancer under propofol-N2O anaesthesia. She had symptoms of dysarthria, bulbar palsy, cerebellar ataxia, Parkinsonism, myosis, pyramidal signs and muscular atrophy of the distal extremities. A cumulative dose-response curve for vecuronium was constructed, and pupillary changes in response to various noxious stimuli were evaluated with concomitant recording of the Spectral-Edge-Frequency 90% (SEF90; the frequency below which 90 percent of the EEG power is located). The dose-response curve for vecuronium and the estimated ED50 value (the 50% blocking dose of vecuronium) in this patient with OPCA were almost identical with those of five ASA I-II patients (27 micrograms.kg-1 vs 31 micrograms.kg-1). The pupil size and the SEF90 did not change after tracheal intubation or surgical stimulation in this patient, while in the control subjects (n = 3), these measures increased in response to both stresses. CONCLUSIONS: The absence of pupillary and SEF90 responses to noxious stimuli suggests a sensitivity to propofol and/or central autonomic dysfunction in patients with OPCA. Although the dose requirement of vecuronium in this patient was similar to that of the control patients, the effects of neuromuscular blockers may vary depending on the severity of muscle atrophy.

Aged↗

PubMed1991

Autonomic stimulation.

Therapeutic stimulation of the autonomic nervous system has been limited by lack of qualitative or quantitative evaluation of autonomic mechanisms. This article provides an historical review of knowledge about autonomic pathways and critical evaluation of available tests of autonomic function. Recent developments in evaluation of autonomic dysfunction and improvement in techniques of neurostimulation have facilitated the development of a number of clinically useful treatments for bladder control, sexual problems, peripheral vascular disease, angina pectoris, and seizure disorders. Future therapeutic measures may allow specific control of hypertension, pain, cardiac arrhythmias, trophic disorders and balance.

Autonomic Nervous System↗

PubMed1989

3,4-Diaminopyridine in the treatment of Lambert-Eaton myasthenic syndrome.

Lambert-Eaton myasthenic syndrome is characterized by muscle weakness, hyporeflexia, and autonomic dysfunction, which result from impaired release of acetylcholine from cholinergic nerve terminals. It is frequently associated with cancer, it is autoimmune-mediated, and treatment has been unsatisfactory. 3,4-Diaminopyridine enhances the release of acetylcholine. In this prospective, double-blind, placebo-controlled crossover study of 12 patients with Lambert-Eaton myasthenic syndrome (7 of whom had cancer), 3,4-diaminopyridine in doses up to 100 mg per day was effective in treating both the motor and the autonomic deficits of the syndrome. Muscle strength increased from an average of 70 percent of normal to 81 percent of normal in the upper extremities, and from 45 to 65 percent of normal in the lower extremities. The amplitudes of compound-muscle-action potentials nearly doubled, increasing from an average of 2.9 mV to 5.0 mV in the arm and from 1.6 mV to 3.1 mV in the leg. Autonomic symptoms were relieved. One patient had a seizure after 10 months of treatment, but other side effects from the drug were minimal and dose-related. We conclude that 3,4-diaminopyridine, either alone or in conjunction with other therapies, may be useful in the treatment of Lambert-Eaton myasthenic syndrome.

4-Aminopyridine↗

PubMed2000

Autonomic effects of radiofrequency catheter ablation.

Radiofrequency (RF) catheter ablation has become the treatment of choice for a variety of supraventricular tachycardias. Autonomic dysfunction may occur during application of RF current; these abnormalities resolve quickly when current delivery is terminated. We present a case of sinus node arrest and AV block in a 69-year-old woman induced by RF catheter ablation of an AV nodal slow pathway. The proposed mechanisms are a Bezold-Jarisch-like phenomenon or paradoxical activation of cardiac C fibres by direct neural sympathetic stimulation or RF-induced myocardial injury. Based on a review of previously published reports, autonomic effects of RF ablation are discussed.

Aged↗

PubMed2000

Cardiovascular dysfunction in multiple sclerosis.

Cardiovascular dysfunction (CD) in multiple sclerosis (MS) is related to involvement of reflex pathways in the brainstem. The battery of CD tests was applied to a group of 40 healthy subjects and 40 patients with MS, divided in 2 subgroups according to the expanded disability status scale (EDSS). The tests included: 1) postural blood pressure changes, 2) postural heart rate changes, 3) heart rate changes on inspiration/forced expiration and 4) ECG R-R interval measurement on the Valsalva maneuver. Both groups were subjected to the functional independence scale (FIM). Imaging studies were reviewed and autonomic dysfunction at other levels was explored. The results showed a statistically significant difference (P < 0.05) in all tests when comparing patients to controls. Tests 1 and 4 had the highest significance, with findings of more severe involvement in patients with a higher EDSS and lower FIM. A correlation was also found between CD and brainstem lesions on MRI (P < 0.01). A significant number of MS patients had evidence of CD. Test 1 may be considered a simple marker, in daily clinical practice, to detect subclinical CD. Subclinical CD is a cause of disability in this group of patients.

Adolescent↗

PubMed1997

Autonomic involvement in multiple sclerosis: a pupillometric study.

To study pupillary autonomic function in multiple sclerosis (MS), we examined 36 subjects with low disability, preserved visual acuity and no recent history (2 years) of optic neuritis or actual visual complaints. Compared to controls, MS patients showed a greater dilatator reaction with darkness and, for the light reflex, a lower amplitude and contraction rate and a greater recovery of pupillary diameter 5 s after the stimulus. Within the MS group, no difference was found comparing patients with or without the following characteristics: nuclear magnetic resonance imaging evidence of midbrain lesions; increased visual evoked potential P100 latency; and a previous history of optic neuritis. No correlation was found between P100 latency, duration of disease and pupillometric parameters. Our results indicate that in MS patients there is autonomic dysfunction with a reduction of parasympathetic tone and a relative increase in sympathetic dilatator tone to the pupils. We suggest that pupillary abnormalities could be due to non-specific impairment of the central pathways subserving pupil functions.

Adaptation, Ocular↗

PubMed2002

Spectral analysis of R-R interval variability by short-term recording in anorexia nervosa.

Subjects with anorexia nervosa (AN) present a number of changes in autonomic system functions, such as thermoregulation, vascular motility, heart rate and rhythm, and blood pressure. We evaluated the changes in the autonomic control of heart rate and blood pressure after postural variation by means of the spectral analysis of R-R interval variability (HRV in 13 female subjects with AN diagnosed on the basis of diagnostic statistical manual (DSM-IV) criteria, a mean age of 25 +/- 5.8 years and a mean body mass index (BMI) of 16.9 +/- 2.6. The controls were 16 healthy female subjects with a mean age of 25 +/- 2.3 years and a normal BMI. The data were statistically evaluated by means of one-way analysis of variance or Student's t test. The high frequency (HF) components of the spectral analysis did not significantly change when passing from clino- to orthostatism in the AN subjects, but there were significant changes in the controls. The changes in the low frequency (LF) components were similar in both groups, but smaller in the AN subjects. However, the difference between the two series was not statistically significant. The variance in the orthostatic R-R intervals recorded in the AN subjects was significantly less than the clinostatic intervals, the intervals recorded in the controls. These results indicate that AN subjects show signs of autonomic dysfunction. The increase in the HF component of the spectral analysis suggests that parasympathetic modulation is abnormally persistent during orthostatism, furthermore, the variability of the R-R intervals indicates that orthosympathetic regulation is also altered in AN.

Adolescent↗

PubMed2006

Cardiovascular and cerebrovascular responses to lower body negative pressure in type 2 diabetic patients.

In diabetic patients, vascular disease and autonomic dysfunction might compromise cerebral autoregulation and contribute to orthostatic intolerance. The aim of our study was to determine whether impaired cerebral autoregulation contributes to orthostatic intolerance during lower body negative pressure in diabetic patients. Thirteen patients with early-stage type 2 diabetes were studied. We continuously recorded RR-interval, mean blood pressure and mean middle cerebral artery blood flow velocity at rest and during lower body negative pressure applied at -20 and -40 mm Hg. Spectral powers of RR-interval, blood pressure and cerebral blood flow velocity were analyzed in the sympathetically mediated low (LF: 0.04-0.15 Hz) and the high (HF: 0.15-0.5 Hz) frequency ranges. Cerebral autoregulation was assessed from the transfer function gain and phase shift between LF oscillations of blood pressure and cerebral blood flow velocity. In the diabetic patients, lower body negative pressure decreased the RR-interval, i.e. increased heart rate, while blood pressure and cerebral blood flow velocity decreased. Transfer function gain and phase shift remained stable. Lower body negative pressure did not induce the normal increase in sympathetically mediated LF-powers of blood pressure and cerebral blood flow velocity in our patients indicating sympathetic dysfunction. The stable phase shift, however, suggests intact cerebral autoregulation. The dying back pathology in diabetic neuropathy may explain an earlier and greater impairment of peripheral vasomotor than cerebrovascular control, thus maintaining cerebral blood flow constant and protecting patients from symptoms of presyncope.

Autonomic Nervous System Diseases↗

PubMed1990

Uraemic autonomic neuropathy.

Symptoms of uraemic autonomic neuropathy are often vague and aspecific. A correct diagnosis of autonomic dysfunction is possible by using several simple and non-invasive tests of parasympathetic and sympathetic control of the cardiovascular system. A defective regulation of the heart rate, due mostly to an afferent lesion, is more common than damage of reflex blood pressure control. The former appears isolated in 14 to 34% of uraemics on haemodialysis and combined to the latter in 18 to 26%. Contradictory results are reported about influence of intermittent haemodialysis, continuous ambulatory peritoneal dialysis and kidney transplantation on disturbed autonomic nervous system. Pathogenesis of uraemic autonomic neuropathy remains unknown and some mechanisms are only putative.

Autonomic Nervous System↗

PubMed2003

[Horner's syndrome associated with Guillain-Barré syndrome].

Autonomic dysfunctions are classically described during the acute phase of Guillain-Barré syndrome (GBS). This involvement concerns visceral afferent, parasympathetic and/or sympathetic efferent fibers and is closely related to sudden deaths in the acute phase of the disease. We report a case of a patient suffering from a GBS associated with a transient Horner syndrome without extraocular muscle involvement. Pharmacological tests of the pupils pointed to an orthosympathetic pre-ganglionic disturbance. Neither clinical nor electrophysiological parameter suggested a broader involvement of the autonomic nervous system.

Acute Disease↗

PubMed1996

Management of diabetic pregnancy complicated by coronary artery disease and neuropathy.

Various manifestations of diabetic neuropathy may complicate pregnancies of young diabetic patients. Of all forms of diabetic neuropathy, autonomic neuropathy, and, in particular, gastropathy, may cause the most devastating complications. Because neuropathy is a common abnormality in young asymptomatic diabetic women, screening for this disorder may be advisable and can be accomplished by relatively simple and noninvasive tests. Screening is best performed before conception or early in pregnancy, because pregnancy itself and its possible complications later modify the autonomic nervous function tests and make testing unreliable. Practitioners and obstetricians who provide care and counseling to young diabetic patients should be familiar with the risks and consequences to maternal and fetal health that may be imposed by the different forms of neuropathy. Moderate-to-severe autonomic dysfunction may be considered a relative contraindication to pregnancy, especially if gastropathy is part of the clinical presentation. The management dilemmas and high mortality and morbidity associated with symptomatic diabetic neuropathy may justify the addition of a new independent class, class N (neuropathy), to the current classification systems for diabetes in pregnancy.

Autonomic Nervous System Diseases↗

PubMed2002

MR guidance of sympathetic nerve blockade: measurement of vasomotor response initial experience in seven patients.

The authors performed sympathetic nerve blockades in seven patients with peripheral ischemia and possible autonomic dysfunction. Magnetic resonance (MR) imaging was used to guide needle placement, to monitor distribution of injected agents, and to measure increases in blood flow, which were as much as 10-fold. MR imaging can provide both procedural imaging guidance and measurement of efficacy for sympathetic nerve blocks.

Adolescent↗

PubMed1987

Hereditary generalized amyloidosis with polyneuropathy. Clinicopathological study of 65 Japanese patients.

A clinicopathological study was made on 65 patients from a small area of Nagano Prefecture, Japan, with hereditary generalized amyloidosis with polyneuropathy to clarify the clinical variety of the disease. Forty-five patients from Ogawa village showed similar clinical features. The age of onset ranged widely from 16 to 62 years. The main neurological manifestations were polyneuropathy starting in the legs and autonomic dysfunction. Lower cranial nerves were also affected in the advanced stages. Severe cardiac and renal involvement was uncommon. All these clinical features are consistent with type I familial amyloid polyneuropathy (FAP). The remaining 20 patients from five unrelated kinships showed unique clinical pictures. Two families from Ogawa village had type I FAP, but 4 out of the 5 affected patients showed marked nephropathy with heavy proteinuria from an early stage. Of the three other families, one, with 10 patients, was notable for the involvement of the central nervous system. Most of the patients showed cerebellar ataxia and pyramidal tract signs in addition to a sensorimotor and autonomic peripheral neuropathy. Another family had 2 siblings who had severe amyloid heart disease from the onset and developed polyneuropathy with autonomic features at an advanced stage. In the third family, onset occurred in the sixth decade in all 3 patients and the course was mild in 2, although the clinical features were those of typical type I FAP. Immunohistochemical study revealed that the amyloid fibril proteins in the patients with all four unusual clinical phenotypes were related to plasma prealbumin. The most common form of hereditary generalized amyloidosis in Japan is type I FAP, but the disease shows considerable variety in the age of onset and involves more systemic organs than previously recognized. The newly recognized clinical forms of hereditary generalized amyloidosis with severe amyloid heart disease or central nervous dysfunction indicate clinical heterogeneity of hereditary amyloidosis with polyneuropathy.

Adolescent↗