Nitrous Oxide-induced Subacute Combined Degeneration Presenting with Dystonia and Pseudoathetosis: A Case Report.

PURPOSE: Nitrous oxide (N2O) is neurotoxic by interfering with vitamin B12 bioavailability. The clinical picture is indistinguishable to that of subacute combined degeneration (SCD). A movement disorder might occur though it is not a characteristic feature. We report a patient with N2O-induced SCD, exhibiting a combination of different involuntary movements. CASE REPORT: A 20-year-old woman presented with one month of progressive unsteady gait, involuntary movements and tingling sensation in a stocking-glove distribution. She had used N2O and ketamine intermittently for recreational purposes for about two years. Neurological examination demonstrated normal cranial nerve functions except for dystonia in the facial muscle and tongue. Her muscle strength was full, but there were bilateral hyperreflexia and extensor plantar response. She exhibited dystonia in four limbs with athetoid movement in fingers and toes, worsened by eye closure. Vibration and proprioception were impaired. Laboratory tests revealed anemia (Hb: 9.9 g/dl) with normal mean corpuscular volume (85.7 fL) and decreased iron level (22 µg/dl) while other results were normal including serum vitamin B12 level (626 pg/ml). Magnetic resonance imaging showed a hyperintense lesion from C1 to C6 level in the posterior column. She was diagnosed as having SCD caused by N2O abuse, presenting with generalized dystonia and pseudoathetosis. The involuntary movements disappeared with vitamin B12 supplementation. CONCLUSION: Movement disorders may be the rare manifestations of SCD associated with N2O abuse. Early recognition of the etiology is vital because it is treatable with vitamin B12 and methionine.

Sympathomimetic syndrome, choreoathetosis, and acute kidney injury following "bath salts" injection.

"Bath salts" is a well known street drug which can cause several cardiovascular and neuropsychiatric symptoms. However, only one case of acute kidney injury has been reported in the literature. We present a case with sympathomimetic syndrome, choreoathetosis, gustatory and olfactory hallucinations, and acute kidney injury following the use of bath salts. A 37-year-old man with past medical history of hypertension and depression was brought to the emergency center with body shaking. Three days before admission he injected 3 doses of bath salts intravenously and felt eye pain with blurry vision followed by a metallic taste, strange smells, profuse sweating, and body shaking. At presentation he had a sympathomimetic syndrome including high blood pressure, tachycardia, tachypnea, and hyperhydrosis with choreoathetotic movements. Laboratory testing revealed leukocytosis and acute kidney injury with a BUN of 95 mg/ dL and a creatinine of 15.2 mg/dL. Creatine kinase was 4,457 IU/dL. Urine drug screen is negative for amphetamine, cannabinoids, and cocaine; blood alcohol level was zero. During his ICU stay he became disoriented and agitated. Supportive treatment with 7.2 liters of intravenous fluid over 3 days, haloperidol, and lorazepam gradually improved his symptoms and his renal failure. Bath salts contain 3,4-methylenedioxypyrovalerone, a psychoactive norepinephrine and dopamine reuptake inhibitor. Choreoathetosis in this patient could be explained through dopaminergic effect of bath salts or uremic encephalopathy. The mechanism for acute kidney injury from bath salts may involve direct drug effects though norepinephrine and dopamine-induced vasoconstriction (renal ischemia), rhabdomyolysis, hyperthermia, and/or volume contraction.

Choreoathetosis in the setting of lithium toxicity.

BACKGROUND: Lithium toxicity has been shown to cause lasting neurological sequelae in certain cases. OBJECTIVE: The authors present a case of choreoathetosis in the aftermath of a presumed episode of lithium toxic reaction. METHOD: The patient was treated by aggressive rehydration; lithium and, ultimately, all psychotropic medication was withheld for a period. RESULTS: The patient showed marked improvement in orientation and movement control; however, some of the choreoathetoid symptoms persisted. CONCLUSION: Patients on combination therapy with lithium and other psychotropics need to be closely monitored for the development of choreoathetoid and other symptoms of overmedication.

Sildenafil induced choreoathetosis in men with Parkinson's disease.

OBJECTIVE: The present report describes a case of choreoathetotic movements which were most probably induced by sildenafil in a patient with Parkinson's disease (PD) treated with levodopa/carbidopa (LD/CD). CASE SUMMARY: A 56-year-old retired man was admitted to hospital because of bizarre, involuntary movements and anxiety. Before admission he had taken sildenafil 100 mg. He had a previous history of PD for 5 years and during the last 3 years he was stable with long-acting LD/CD and selegiline. He is in Stage 2 according to Hoehn and Yahr Staging of PD. The patient did not have any problems with erectile function and he took sildenafil 50 minutes after the last daily dose of LD/CD. The patient was discharged from the hospital 12 hours after the admittance without any symptoms of choreoathetosis. CONCLUSION: Choreoathetotic dyskinesia is an adverse effect which was provoked by sildenafil administration (drug abuse) in a previously stabile responder to LD therapy, but probably had a lower threshold for dyskinesia. Predisposition for this pharmacokinetic interaction could be a short time interval between LD and sildenafil applied in high dosage.

Cisatracurium in "weakening doses" assists in weaning from sedation and withdrawal following extended use of inhaled isoflurane.

OBJECTIVE: Isoflurane was used to treat a patient with status asthmaticus refractive to standard therapeutic measures. The patient developed a significant withdrawal syndrome when the isoflurane was weaned. A case is reported here where this withdrawal syndrome was treated successfully by using a weakening dose neuromuscular blockade with cisatracurium. DESIGN: Case report. SETTING: Pediatric critical care unit. PATIENT: A 4-yr-old girl with severe reactive airways disease. INTERVENTIONS: The use of weakening doses of cisatracurium to assist in weaning from mechanical ventilation in the setting of withdrawal symptoms following the extended use of inhaled isoflurane. MEASUREMENTS AND MAIN RESULTS: Despite treatment with mechanical ventilation, intravenous corticosteroids, and bronchodilators for status asthmaticus, the patient required inhaled isoflurane. She became tolerant to isoflurane over an extended period of time; her tolerance was associated with a specific withdrawal syndrome, with the development of choreoathetoid movements resulting in poor pulmonary coordination and agitation. Conventional medical treatment of withdrawal failed. Finally, by using an infusion of cisatracurium at weakening doses to assist in the control of these choreoathetoid movements, the isoflurane and ventilator support were weaned. CONCLUSIONS: Weakening doses of cisatracurium may be used safely to control unpleasant motor symptoms secondary to tolerance of isoflurane. This may have a use in other circumstances where agitation in mechanically ventilated patients is not due to pain or anxiety.

Choreoathetosis - an unusual adverse effect of dihydroartemisinin-piperaquine: a case report.

BACKGROUND: Dihydroartemisinin-piperaquine is a combination of dihydroartemisinin and piperaquine which is highly effective in the treatment of uncomplicated falciparum malaria. Its adverse effects are generally tolerable and temporary. Choreoathetosis, an involuntary movement disorder characterized by continuous irregular twisting of the body, is not a documented adverse effect of this medication. CASE PRESENTATION: A 41-year-old Cameroonian man of black African ethnicity was brought to our primary care hospital because over the previous 6 hours he had been experiencing involuntary twisting movements of his body and he no longer had control of his limbs. Earlier that day, he had been prescribed an appropriate dose of dihydroartemisinin-piperaquine in our hospital. The abnormal movements started approximately 3 hours after ingesting the first dose of the drug. The review of systems and his past history were unremarkable. On clinical examination, he was conscious and oriented but was unsteady and displayed continuous generalized irregular twisting movements combined with abrupt low amplitude flinging of his limbs. Dihydroartemisinin-piperaquine-induced generalized choreoathetosis was diagnosed. He was sedated with diazepam and dihydroartemisinin-piperaquine was discontinued. The antimalarial drug was substituted with artemether-lumefantrine combination. The clinical progress was good and he was discharged home after 72 hours. No further abnormalities were noted during 7 months of follow-up. CONCLUSION: Although dihydroartemisinin-piperaquine is increasingly popular as a well-tolerated/efficacious antimalarial drug, clinicians must note the rare possibility of choreoathetosis as an adverse effect of this medication and educate patients accordingly.

Psychosis and Wilson's disease: a case report.

In this article we present a case of a 26-year-old woman with clinical picture of acute psychosis, as the first and main manifestation of Wilson's disease, who developed abnormal involuntary choreoathetoid limb movements, few days after initiation of neuroleptic therapy. At the first movement neurological symptoms were misinterpreted as side effect of haloperidol, but consulted neurologist suggested additional diagnostic procedure which confirmed Wilson's disease. Psychiatric symptomatology and abnormal involuntary movements were the clinical manifestation of this disease, which improved with neuroleptic and chelating treatment. Interdisciplinary approach with good collaboration of psychiatrists and neurologists is crucial for Wilson's disease, because early diagnosis and treatment without delay is critical to the prognosis. This case serves as a reminder that involuntary movements can be side effect of antipsychotics but also the clinical manifestation of some illnesses, for example Wilson's, Huntington's and Fuhr's diseases.

Reversal of tricyclic-overdosage-induced central anticholinergic syndrome by physostigmine.

Although there is evidence that the delirium, stupor, coma, and seizure-like activity seen in overdosage with tricyclic antidepressants and antiparkinson drugs are due to the central anticholinergic activity of these agents, patients with overdosage of these drugs are still frequently misdiagnosed. The authors present a case of reversal of anticholinergic-drug-induced prolonged coma, myoclonus, and choreoathetosis by physostigmine. This report supports the anticholinergic basis of the clinical manifestations of such overdosages, provides information on the role of acetylcholine and dopamine in psychiatric and movement disorders, and illustrates dramatically the need for accurate diagnosis and treatment.

"On-off" syndrome treated with lithium carbonate: a case report.

The authors describe a 42-year-old man who developed "on-off" episodes and for whom lithium carbonate proved an effective treatment. They speculate that the predominant effect of lithium in the "on-off" syndrome is to prevent L-dopa desensitization of the dopamine receptor.

Choreoathetosis as a side effect of gabapentin therapy in severely neurologically impaired patients.

OBJECTIVE: To evaluate the relationship of gabapentin therapy with choreoathetotic movements in mentally retarded patients treated with intractable epilepsy. DESIGN: Case reports of 2 institutionalized patients who developed choreoathetosis temporally related to adjunctive therapy with gabapentin at dosages of 1200 to 1800 mg/d. RESULTS: Both patients experienced resolution of abnormal movements on discontinuation of the therapy. One patient developed recurrent choreiform movements after drug rechallenge. CONCLUSION: We suggest that, in patients with mental retardation and epilepsy, involuntary movements may either occur as reversible side effects of gabapentin therapy or result from a previously undescribed adverse drug interaction with other antiepileptic agents.

Delayed choreoathetosis following acute carbon monoxide poisoning.

Ten days after accidental exposure to carbon monoxide, a 17-year-old youth developed transitory choreoathetosis of both arms, face, and neck, with moderate dysarthria. CT revealed symmetric bilateral infarction in the head of the caudate nucleus, the putamen, and the small parts of the anterolateral globus pallidus.

Effect of age at onset on progression and mortality in Parkinson's disease.

We examined longitudinal disability scores in 54 patients with Parkinson's disease followed for 6 years at UCLA. We sorted data into 3 groups based on age at onset of symptoms: group A, onset under 50 years; group B, 50 to 59 years; group C, 60 years or older. There were no significant differences between groups initially. All 3 groups improved dramatically when levodopa was given, but group A showed significantly less disability in years 4, 5, and 6 than did group C. The groups did not differ with respect to side effects. To determine if age at onset affected mortality, we sorted records from 4 geographically diverse centers into the same 3 groups. Results on 359 patients followed for 3,314 person-years, covering a period of 17 years after onset of symptoms, showed that group A had the most favorable observed-to-expected mortality ratio, 1.82, compared with 2.17 and 2.20 for groups B and C respectively, but the difference was not statistically significant. Results from the disability analyses indicate that patients with onset of Parkinson's disease under 50 years of age may have a more favorable prognosis than those whose symptoms begin in later years.

Phenytoin-induced dystonia and choreoathetosis in two retarded epileptic children.

Similar movement disorders developed in two 8-year-old retarded children while they were receiving phenytoin. Seizures subsequent to a diphtheria-pertussis-tetanus immunization had developed in each child at 1 to 2 months of age. A static encephalopathy ensued, characterized by mental retardation, ataxia, spasticity, and a mixed seizure disorder. Intermittent dystonia and choreoathetosis developed insidiously while serum phenytoin concentrations were in the therapeutic range. Sustained dystonia and choreoatheosis developed 2 hours after an oral provocation with phenytoin. The baseline abnormalities on the electroencephalogram remained unchanged during the choreoathetosis. Recognizable metabolic abnormalities known to be associated with similar movement disorders were excluded. It was concluded from these studies that the movement disorder is secondary to phenytoin and can occur at therapeutic serum concentrations. Phenytoin is a central anticholinergic agent and a central stimulant of serotonin, and may induce movement disorders as a result of altering these neurotransmitters in the brain. The variable expression of these movement disorders may relate to the nature of the preexisting striatal insult.

Phenytoin-induced choreoathetosis in infancy: case reports and a review.

Three cases of choreoathetosis which developed during phenytoin therapy in children less than 2 years of age are described. The most striking clinical manifestations included the sudden onset of restlessness and agitation with superimposed choreoathetosis. None of these children had toxic levels of phenytoin in the blood. Discontinuation of phenytoin resulted in prompt cessation of the symptoms. Phenytoin-induced choreoathetosis should be a diagnostic consideration in children with a preexisting CNS insult who manifest violent choreoathetosis during therapy for seizure control. This consideration is especially pertinent in the pediatric intensive care unit, where other more common causes of agitation could be misdiagnosed.

d-Amphetamine-induced "floating limb" syndrome in young rhesus monkeys.

Acute d-amphetamine administration to young rhesus monkeys (N = 10) caused a motor syndrome of hypoactivity and chorea-like postures and motor movements which we have termed "floating limb". Frequently after subcutaneous injections of 0.3 or 0.6 mg/kg d-amphetamine, an affected monkey raised one or both legs or arms and held the limb(s) motionless in the air. Affected limbs were usually returned to a normal position if they appeared to enter the animal's visual field. In other cases, the monkey assumed bizarre and contorted postures which were held for prolonged periods. Such postures were often accompanied by gentle repetitive brushing of the ears and facial hair with extremities of the affected limbs. Quantification of the frequency of these movements showed that they occurred regularly for 90-150 min after d-amphetamine. Hydroxyamphetamine, a peripherally-acting amphetamine analog, did not induce floating limb, indicating that the behavior was probably mediated by central actions of d-amphetamine. A similar disorder has been reported occasionally in other studies with monkeys and cats. It may be related to the chorea that is seen in humans after the use of amphetamine and other stimulants. d-Amphetamine treatment in young monkeys may provide a viable model of human choreoathetoid disorders induced by disease or drug use.

Experimental choreoathetosis produced by injection of a gamma-aminobutyric acid antagonist into the lentiform nucleus in the monkey.

An experiment is reported in which injections of the gamma-aminobutyric acid antagonist bicuculline into the lentiform nucleus of the monkey gave rise to a contralateral choreoathetoid dyskinesia. Evidence was obtained of functional heterogeneity between the dorsal and ventral lentiform nucleus, in that only injections into its ventral part gave rise to dyskinesia.

Midazolam-induced athetoid movements of the lower extremities during epidural anesthesia reversed by physostigmine.

Midazolam is a short-acting, water-soluble benzodiazepine used for induction and maintenance of general anesthesia and as an adjunct to regional anesthesia. This substance produces several types of untoward reactions, including agitated excitement, mental confusion, and uncooperativeness, as well as dystonic extrapyramidal reactions, such as tonic clonic movements, muscle tremor, and athetoid movements. We describe two patients who developed akinesthesia with athetoid movements of the lower extremities after receiving midazolam as a premedication and as an adjunct to epidural anesthesia. These movements occurred with a sensory level of T4 as assessed by pinprick, even though the patients were unable to move their lower extremities.

[Tardive dyskinesia induced by neuroleptics. A review of the literature]. / Les dyskinésies tardives des neuroleptiques. Revue de la littérature.

Neuroleptics can induce not only early but also tardive extrapyramidal side effects. The former were described as early as in the beginning of the therapeutic era; the latter, known as tardive dyskinesia, are essentially made of bucco-linguomasticatory dyskinesia, sometimes accompanied by other extrapyramidal symptoms, among others choreo-athetoid movements of the limbs. This complication was most often reported in elderly people, when a long-lasting neuroleptic treatment is withdrawn; but it can also appear in young people and after a neuroleptic treatment of only a few weeks duration. The trouble occurs or is worsened when neuroleptics are withdrawn and is reduced when dosage is increased; it responds to the theoretical model of denervation supersensitivity. Symptoms may be reduced by blocking the dopaminergic receptor, or by reducing dopaminergic transmission, or, may be, by increasing cholinergic activity. Now, treatment is first and foremost prevention.

[Choreoathetotic movements in a patient with epilepsy after treatment with diphenylhydantoin]. / Wystepowanie ruchów plasawiczo-atetotycznych u chorego na padaczke w nastepstwie leczenia difenylhydantoina.

The authors describe a very rare case of choreoathetotic movements which appeared in an epileptic in strict relationship to a toxic concentration of diphenylhydantoin in the serum. These movements were reversible and disappeared after reduction of drug dose and its serum concentration falling to the therapeutic range.