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PubMed2003

Acute femoral neuropathy secondary to an iliacus muscle hematoma.

We present a patient with a spontaneous iliacus muscle hematoma, appearing immediately after a minor physical maneuver, presenting with pain and femoral neuropathy initially evidenced by massive quadriceps muscle fasciculations. A magnetic resonance imaging (MRI) study of the pelvic area confirmed the diagnosis, showing a hematoma secondary to a partial muscle tear. The patient was managed conservatively, and the continuous muscle activity ceased in 3 days, with progressive improvement of the pain and weakness. The recovery was complete. Femoral neuropathy is uncommon and usually due to compression from psoas muscle mass lesions of diverse nature, including hematomas. Usually subacute, femoral neuropathy may present acutely in cases of large or strategically placed compressive femoral nerve lesions, and may require surgical evacuation. The case presented herein is remarkable since the muscle hematoma appeared after a nonviolent maneuver, fasciculations were present at onset, and conservative management was sufficient for a full recovery.

Acute Disease↗
PubMed1998

Clinical neurophysiology in the diagnosis of amyotrophic lateral sclerosis: the Lambert and the El Escorial criteria.

For many years, the only published criteria for the electrodiagnostic (EDX) recognition of amyotrophic lateral sclerosis (ALS) were those formulated by Lambert (1957; 1969). In 1990, different EDX guidelines were incorporated in the all-inclusive diagnostic criteria formulated by a subcommittee on ALS of the World Federation of Neurology, which met in El Escorial, Spain. Unfortunately, particularly in regard to the EDX requirements, the 'El Escorial criteria' have several flaws which compromise their usefulness. These include: (1) they ignore the fact that whenever upper and lower motor neuron disorders co-exist, as they characteristically do with ALS, the motor unit potential firing pattern is controlled by the upper motor neuron lesion; (2) they markedly devalue the usefulness of detecting fasciculations and, through presumably typographical error, state that the 'absence' rather than the 'presence' of fasciculations supports the diagnosis of ALS; this view is in direct conflict with the opinions expressed by most electromyographers; (3) they contain a statement regarding how the diagnosis of ALS is confirmed by the EDX studies which is confusing and, for two of the body regions (bulbar; thoracic), unrealistic; (4) finally, many of the EDX features they listed supporting the recognition of possible LMN degeneration appear to be mislabeled, while a few features in the EDX criteria are incorrect.

Amyotrophic Lateral Sclerosis↗
PubMed2004

Neuromyotonia.

There is increasing evidence that autoimmunity is implicated in the pathogenesis of peripheral nerve hyperexcitability (neuromyotonia, NMT and Cramp-fasciculation syndrome C-FS ) and in Maladie de Morvan in which CNS features are also present. All three conditions can associate with thymoma, myasthenia gravis and other autoimmune disorders, and can often respond to plasma exchange. In NMT, patient's plasma or IgG can transfer the electrophysiological features to mice, and can reduce voltage-gated potassium channel currents in vitro. Antibodies to voltage-gated potassium channels can be detected in the serum of many patients who have peripheral nerve hyperexcitability, and also in those with Maladie de Morvan. These latter patients have clinical features similar to limbic encephalitis in which VGKC antibodies can also occur. Thus neuromyotonia, cramp-fasciculation syndrome and Maladie de Morvan can occur as antibody-mediated autoimmune ion channelopathies like myasthenia gravis and the Lambert-Eaton myasthenic syndrome. These discoveries should aid diagnosis and offer new approaches to treatment.

Animals↗
PubMed2000

Spin trapping agent phenyl-N-tert-butylnitrone prevents diisopropylphosphorofluoridate-induced excitotoxicity in skeletal muscle of the rat.

Indirect evidence suggests that reactive oxygen species (ROS) may mediate muscle fiber necrosis following muscle hyperactivity induced by the anticholinesterase diisopropylphosphorofluoridate (DFP). Pronounced muscle fasciculations and muscle fiber necrosis were seen when acetylcholinesterase (AChE) activity was reduced to less than 30% of control. The spin trapping agent phenyl-N-tert-butylnitrone (PBN) was used in vivo to directly assess the formation of ROS during DFP (1.75 mg/kg, s.c.) induced muscle hyperactivity. Pretreatment with PBN (300 mg/kg, i.p.), the concentration necessary for in vivo spin trapping, prevented muscle hyperactivity as well as necrosis and attenuated the DFP induced AChE inhibition otherwise seen in DFP only treated rats. PBN had no effect when given after fasciculations were established. Muscle extracts from PBN and DFP treated rats subjected to electron spin resonance (ESR) spectroscopy tested negative for ROS. While the role of PBN as an antioxidant is well established, its prophylactic effect against excitotoxity induced by an AChE inhibitor are due to its protection of AChE, an unexpected non-antioxidant action.

Acetylcholinesterase↗
PubMed1999

Fibrillation and sharp-waves: do we need them to diagnose ALS?

BACKGROUND: Fibrillation/sharp-waves (fibs-sw) are considered an essential neurophysiological marker in Amyotrophic Lateral Sclerosis (ALS). We investigated how frequently an initial electrophysiological (EMG) evaluation, performed in ALS patients, revealed no fibs-sw. METHODS: The initial EMG studies from 68 ALS patients were analysed. The presence of fibs-sw in at least two muscles of each limb and two cranial-innervated muscles was assessed. RESULTS: We observed no fibs-sw in cranial-innervated muscles, and 7/15 bulbar-onset patients had no fibs-sw in limb muscles. Two of 28 upper-limb-onset patients, who had a short history before EMG, showed no fibs-sw. All lower-limb-onset patients had fibs-sw. Generally patients with no fibs-sw had diffuse abundant fasciculations. CONCLUSIONS: The absence of fibs-sw should not necessarily preclude the neurophysiological diagnosis of ALS in bulbar-onset patients. Early affected upper-limb-onset patients might show no fibs-sw, but profuse fasciculations associated with mild to moderate denervation-reinnervation in weak muscles are found.

Adult↗
PubMed2006

Altered axonal excitability properties in amyotrophic lateral sclerosis: impaired potassium channel function related to disease stage.

Fasciculations are a characteristic feature of amyotrophic lateral sclerosis (ALS), and can arise proximally or distally in the motor neuron, indicating a widespread disturbance in membrane excitability. Previous studies of axonal excitability properties (i.e. threshold electrotonus, strength-duration time constant) have suggested respectively that change in potassium or sodium channels may be involved. To reinvestigate these changes and explore their correlation with disease stage, multiple axonal excitability properties (threshold electrotonus, strength-duration time constant, recovery cycle and current-threshold relationship) were measured for the median nerve at the wrist in 58 ALS patients, and compared with 25 age-matched controls. In ALS, there were greater changes in depolarizing threshold electrotonus (i.e. less accommodation) (P < 0.001) and greater supernormality in the recovery cycles (P < 0.001). These abnormalities were more prominent in patients with moderately reduced CMAP (1-5 mV). Modelling the excitability changes in this group supported the hypothesis that axonal potassium conductances are reduced, resulting in increased supernormality despite membrane depolarization. The tendency for strength-duration time constant to be prolonged in ALS was only significant for patients with normal CMAP amplitude (>5 mV). Patients with severely reduced CMAP (<1 mV) alone showed reduced threshold changes to hyperpolarizing current. These results suggest a changing pattern of abnormal membrane properties with disease progression. First, persistent Na+ conductance increases, possibly associated with collateral sprouting, and then K(+) conductances decline. Both changes cause axonal hyperexcitability, and may contribute to the generation of fasciculations. These serial changes in axonal properties could provide insights into the pathophysiology of ALS, and implications for future therapeutic options.

Adult↗
PubMed1979

Dantrolene and suxamethonium. The effect of pre-operative dantrolene on the action of suxamethonium.

A single oral dose of dantrolene (100--150 mg) given at least 2 hr pre-operatively has been found to reduce significantly the strength of muscular fasciculations, the hyperkalaemia and the incidence of muscle pains following suxamethonium (from 56 to 4%) in a series of forty-eight patients compared with controls. The biceps EMG of the fasciculations was unchanged by dantrolene; the incidence of troublesome side effects was low (9%) and there did not appear to be any alteration in the duration of action of suxamethonium.

Adult↗
PubMed1995

Effects of magnesium sulphate on suxamethonium-induced complications during rapid-sequence induction of anaesthesia.

Twenty patients were studied in a double-blind manner to investigate whether magnesium sulphate, when given during a rapid-sequence induction of anaesthesia, lessens the side effects caused by suxamethonium. Patients were randomly allocated to two groups; equal volumes of either magnesium sulphate (40 mg.kg-1) or saline were given during rapid-sequence induction of anaesthesia, after thiopentone but before the administration of suxamethonium (1.5 mg.kg-1). The changes in the serum potassium concentration, the degree of muscle fasciculations and the presence of postoperative myalgia were recorded. The mean serum potassium concentration increased by 0.08 mmol.l-1 in the magnesium group and by 0.1 mmol.l-1 in the control group at 2 min after injection of suxamethonium; in neither group was there a significant increase from baseline values. The systolic blood pressure and heart rate increased in both groups after tracheal intubation. The incidence of fasciculations was significantly lower in the magnesium group. Magnesium did not clinically prolong muscle relaxation. There was no difference between the groups in the incidence of myalgia after surgery (one patient in each group). Since no significant increase in the serum potassium concentration was demonstrated, no assessment could be made of the effect of magnesium sulphate on the serum potassium concentration after administration of suxamethonium.

Adolescent↗
PubMed1990

Effects of precurarisation on suxamethonium-induced postoperative myalgia during the first trimester of pregnancy.

Two hundred and fifty women undergoing termination of pregnancy during the first trimester under general anaesthesia were studied to determine the effects of precurarisation on suxamethonium-induced postoperative myalgia and on the need for postoperative analgesics after suxamethonium. Either alcuronium (0.03 mg/kg), atracurium (0.04 mg/kg), tubocurarine (0.05 mg/kg), vecuronium (0.01 mg/kg) or saline was administered in a double-blind manner 4 min before giving suxamethonium. An additional 50 patients were studied who received isoflurane rather than precurarisation and suxamethonium. Every pretreatment prevented fasciculations better than did saline (P less than 0.001). In the saline group, 92% of patients had fasciculations and in the other groups this ranged from 8 to 32%, respectively. On the first postoperative day, 76% of the patients in the saline group had myalgia while myalgia was manifested in 28, 54 and 34% of patients given alcuronium, tubocurarine or vecuronium, respectively (P less than 0.05). Atracurium failed in this effect with 62% having myalgia. In the isoflurane group, none of the patients complained of myalgia on the first postoperative morning. The need for analgesics was less (P less than 0.005) in the isoflurane group (8%) and in the pretreatment groups (18-27%) than in the saline group (42%). It is concluded that precurarisation with tubocurarine, vecuronium or, most effectively, with alcuronium but not with atracurium decreases suxamethonium-induced postoperative myalgia and seems to be necessary also during the first trimester of pregnancy.

Abortion, Induced↗
PubMed1983

Abstinence syndrome from therapeutic doses of oxazepam.

A patient developed severe anxiety, moodswing, depression, and thinking disorder 24 hours after abruptly stopping oxazepam, of which he had taken 30 mg 3 times a day for two months, for anxiety and panic attacks. Oxazepam was restarted and tapered off gradually; nevertheless, a relatively severe abstinence syndrome occurred. Muscular fasciculations and moodswing were very marked. The patient also experienced significant anxiety, depression, moodswing, and muscular fasciculations for two months after detoxication from oxazepam. Subsequently, the patient's panic attacks were treated successfully with propranolol hydrochloride. This report concludes with a brief review of the literature on benzodiazepine withdrawal.

Adult↗
PubMedDate not supplied

Neuromuscular hyperexcitability features in patients suffering from musculoskeletal pain: a neuroepidemiologic survey.

We studied the reported frequencies of clinical complaints of neuromuscular hyperexcitability (muscle cramps and fasciculations) in random samples of 527 Dutch adults, who were and 253 Dutch adults, who were not suffering from musculoskeletal pain and tenderness. Data were collected by telephone-interview and by self-administered questionnaire. Muscle cramps and fasciculations were recorded more frequently in the category that suffered from musculoskeletal pain (p less than 0.001). This association warrants further investigation into the possible intrinsic role of neuromuscular hyperexcitability in musculoskeletal pain and primary fibromyalgia.

Cross-Sectional Studies↗
PubMed1991

Botulinum toxin in the treatment of facial dyskinesias.

Patients with hemifacial spasm (N = 25), blepharospasm (n = 8), and benign eyelid fasciculation (n = 2) were treated with botulinum toxin injections (PHLS, Porton Down, England). All patients reported substantial symptomatic relief. Marked improvement was seen in fifteen patients with hemifacial spasm and six patients with blepharospasm. Benign eyelid fasciculation was completely abolished. Beneficial effects was evident two to three days after injections, became maximum at one week, and remained effective for up to six months. Side effects were transitory and mild. They included periorbital edema, mild diplopia, ptosis and facial weakness. Only in two patients was ptosis unacceptable. Severity of side effects was dose-related. Reinjections had similar efficacy. Botulinum toxin therapy is a safe and effective treatment for these facial dyskinesias and should be considered a viable alternative to surgical procedures.

Adult↗
PubMed1988

[Post-radiation brachial plexopathy. Persistent conduction block. Myokymic discharges and cramps].

The distinction between radiation and tumor brachial plexopathy may be difficult. The electrophysiological recording of myokymic discharges, frequently present in the former but rare in the latter type of plexopathy, can be helpful for the diagnosis. However, the pathophysiology and the site of origin of these discharges remain unclear. We describe a patient presenting with radiation brachial plexopathy, clinical myokymia, cramps and pain. In this patient, the myokymia--due to abundant myokymic discharges--and the cramps, were related to the existence of persistent conduction block of several years duration. Several findings suggest that the myokymic discharges were generated on blocked axons: voluntary activity did not influence their occurrence nor modify their course; the motor unit potentials involved in the discharges were not evoked by stimulation proximal to the site of the conduction block, whereas the stimulation distal to this site could evoke, modify the rhythm, or interrupt the course of the discharges; the latency of these evoked responses indicated that the site of reflection was proximal on the axon, and likely coincided with that of the conduction block. Recent observations (Roth and Magistris, 1987b) indicated that myokymia, produced by numerous single or grouped fasciculations generated on axon terminals, may be related to persistent conduction blocks of various etiologies. The present case demonstrates that myokymia provoked by myokymic discharges may as well be related to persistent conduction block. The reason why these blocks are accompanied by fasciculations in some situations and by myokymic discharges in others remains an unsolved question. The cramps observed in this patient were also of interest as they occurred in the muscle territory of blocked axons and were provoked by passive muscle shortening. Their origin, distal to the conduction block, is unknown. Finally, a neurolysis did not prevent the progressive transformation of conduction block into axonotmesis.

Brachial Plexus↗
PubMed1987

["Self-taming": an alternative to the prevention of succinylcholine-induced pain].

Muscle pain associated with single-bolus administration of suxamethonium is reported to be one of the common complications of this technique. Since suxamethonium is the most commonly used relaxant in our department and priming with nondepolarizing muscle relaxants is also reported to be linked with complications, while the literature concerning this problem is very contradictory, we wondered if the so-called "self-taming" method represents an alternative to pretreatment with nondepolarizing muscle relaxants. One hundred thirty-two patients (69 male, 63 female) were randomly allocated to three groups. Anesthesia was induced with thiopentone 7 mg/kg body weight. Group 1 (n = 44) was pretreated with 2 mg pancuronium bromide 3 min prior to full relaxation with suxamethonium 1.5 mg/kg. Group 2 (n = 43) received no pretreatment. Group 3 (n = 45) received 4 mg suxamethonium i.v. after induction. One minute later the remaining dose of suxamethonium was applied ("self-taming"). Muscle fasciculation and postoperative myalgia were verified by means of a score. Neuromuscular transmission was recorded on a monitor after controlled train-of-four stimulus and time of onset of neuromuscular blockade was measured. With regard to muscle fasciculation, postoperative pain, and onset of neuromuscular blockade, "self-taming" with suxamethonium yielded results identical to pretreatment with pancuronium bromide. It may therefore be considered as an alternative to pretreatment with nondepolarizing muscle relaxants.

Adult↗
PubMed1981

Biphasic change in serum potassium concentration following a single dose of succinylcholine chloride.

Serum potassium concentration during the thiopentone-succinylcholine induction of endotracheal anesthesia was examined in 53 patients undergoing elective surgery. There was no change in serum potassium after thiopentone. During muscle fasciculation serum potassium fell by 0.37 meq/L and returned to the initial level by the end of fasciculation. During muscle relaxation there was a further rise of 0.41 meq/L. The expected increase in serum potassium following the administration of succinylcholine is shown to be preceded by a transient decrease.

Adolescent↗
PubMed1998

Neurolin, the goldfish homolog of DM-GRASP, is involved in retinal axon pathfinding to the optic disk.

Young axons of new retinal ganglion cells (RGCs) in the continuously growing goldfish retina fasciculate with one another and their immediate forerunners on their path toward the optic disk and along the optic nerve. They express the immunoglobulin superfamily cell adhesion molecules (CAMs) neurolin (DM-GRASP) and the L1-like E587 antigen. Repeated injections of Fab fragments from polyclonal antisera against neurolin (neurolin Fabs) into the eye of 3. 4-cm-long and rapidly growing goldfish caused highly aberrant pathways of young RGC axon subfascicles in the dorsal retina. Many axons grew in circles and failed to reach the optic disk. In contrast, E587 Fabs, used in parallel experiments, disrupted the fascicles but did not interfere with the disk-directed growth. Neurolin Fabs also disturbed axonal fasciculation in vivo as well as in vitro but less severely than E587 Fabs. Coinjections of both Fabs increased defasciculation of the dorsal axons in both aberrant and disk-directed routes. They also disrupted the order of young RGC axons in the optic nerve more severely than E587 Fabs alone. This demonstrates that the development of tight and orderly fascicles in the dorsal retina and in the optic nerve requires both E587 antigen and neurolin. More importantly, our results suggest an involvement of neurolin in RGC axonal guidance from the retinal periphery to the optic disk. Because disrupted fascicles and errant axon routes were found only in the dorsal retinal half, a cooperation with so-called positional markers may be conceived.

Activated-Leukocyte Cell Adhesion Molecule↗
PubMed2000

Amyotrophy in prion diseases.

Amyotrophic lateral sclerosis was once thought to be caused by persistent viral infection, partly because some patients with transmissible Creutzfeldt-Jakob disease showed prominent amyotrophy. However, in the past 15 years there has been little interest in the amyotrophy in prion diseases, and the possible link to amyotrophic lateral sclerosis has been eschewed. We analyzed case reports of prion disease published after 1968 for evidence of amyotrophy. We defined amyotrophy as clinically evident fasciculation buttressed by electromyographic results in some cases. We sought evidence of motor neuron degeneration at autopsy. Prion disease was proved by transmissibility, immunohistochemistry demonstration of protease-resistant prion protein, or finding a mutation in the prion protein gene. Amyotrophy was noted in 27 patients: 13 with sporadic Creutzfeldt-Jakob disease, 2 with familial Creutzfeldt-Jakob disease, and 12 with Gerstmann-Sträussler-Scheinker disease. Of the 27, 23 showed clinical fasciculation and 10 had electromyographic evidence of denervation. The spinal cord was examined in 8 patients: 6 showed loss of motor neurons, 1 showed vacuolation of motor neurons, and 1 reported no abnormalities. Another 23 patients had typical histopathological characteristics but lacked molecular or biochemical proof of prion disease. The total number of patients with amyotrophy and proven prion disease that we identified was 50. This case review supports the belief that amyotrophy is occasionally a prominent feature of Creutzfeldt-Jakob disease and underscores the importance of documenting lower motor neuron function and the crucial role of examining the spinal cord at autopsy in cases of prion disease.

Amyotrophic Lateral Sclerosis↗
PubMed2001

Neurologic findings in Machado-Joseph disease: relation with disease duration, subtypes, and (CAG)n.

CONTEXT: Machado-Joseph disease (MJD), an autosomal dominant spinocerebellar degeneration caused by an expanded CAG repeat on chromosome 14q32.1, is a heterogeneous disorder for clinical manifestations. The reasons for the wide range of neurologic findings in this disease are poorly understood. OBJECTIVE: To explain part of this heterogeneity through the association of the neurologic findings with sex, disease duration, age of onset, clinical type, and size of CAG repeat expansion. DESIGN: A case-control study. SETTING: Ambulatory care. PATIENTS: A consecutive sample of 62 patients with MJD. MAIN OUTCOME MEASURE: Neurologic signs. RESULTS: A direct relationship was found between the disease duration and severity of gait and limb ataxia, dysarthria, dysphagia, fasciculations, pyramidal syndrome, and ophthalmoplegia (P<.02). The most severe forms of nuclear ophthalmoplegia were associated with type 1 MJD, whereas those of supranuclear ophthalmoplegia were associated with type 3 MJD (P<.001). It was also found that higher mean (CAG)(n) lengths were associated with worse degrees of the pyramidal syndrome and dystonia (P<.001). The presence and severity of nystagmus, eyelid retraction, rigidity and/or bradykinesia, and optic atrophy were not clearly associated with any of the predictive variables under study. CONCLUSIONS: Disease duration can explain part of the heterogeneity of ataxia, dysarthria, dysphagia, fasciculations, pyramidal syndrome, and ophthalmoplegia, in MJD. Type 1 MJD was positively associated with nuclear ophthalmoplegia; type 3 MJD was positively associated with supranuclear ophthalmoplegia. Higher mean CAG lengths were found to correlate with the pyramidal syndrome and dystonia. Nystagmus, eyelid retraction, rigidity and/or bradykinesia, and optic atrophy were hardly attributable to any known reason or variable.

Adult↗