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.

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.

Reversible choreoathetosis after the administration of ceftriaxone sodium in patients with end-stage renal disease.

Neurologic manifestations, such as myoclonus, asterixis, seizures and altered level of consciousness, may be induced in patients with impaired renal function receiving ß-lactam antibiotics, which stem in part from drug accumulation because of altered pharmacokinetics. Because of its long half-life and easy penetration into the cerebrospinal fluid, the third generation cephalosporin, ceftriaxone (CTRX), is often chosen to treat patients with end-stage renal disease (ESRD). Here, the authors describe 4 patients with ESRD complicated with bacterial infection and choreoathetosis after the administration of CTRX. Choreoathetosis disappeared without leaving sequelae after CTRX therapy was withdrawn, although the severity and symptom duration varied. To our knowledge, there are few reports on choreoathetosis associated with ß-lactam antibiotic administration in patients with kidney diseases. To prevent delayed diagnosis, one should bear in mind that choreoathetosis might occur in patients with ESRD treated with CTRX, when it is given in high or even regular doses.

Age, dose, and time-dependency of plasma and tissue distribution of deltamethrin in immature rats.

The major objective of this project was to characterize the systemic disposition of the pyrethroid, deltamethrin (DLT), in immature rats, with emphasis on the age dependence of target organ (brain) dosimetry. Postnatal day (PND) 10, 21, and 40 male Sprague-Dawley rats received 0.4, 2, or 10 mg DLT/kg by gavage in glycerol formal. Serial plasma, brain, fat, liver, and skeletal muscle samples were collected for up to 510 h and analyzed for DLT and/or 3-phenoxybenzoic acid (PBA) content by high-performance liquid chromatography. Toxicokinetic data from previous experiments of the same design with young adult (PND 90) rats (Kim, K.-B., Anand, S. S., Kim, H. J., White, C. A., and Bruckner, J. V. [2008]. Toxicokinetics and tissue distribution of deltamethrin in adult Sprague-Dawley rats. Toxicol. Sci. 101, 197-205) were used to compare to immature rat data. Plasma and tissue DLT levels were inversely related to age. Preweanlings and weanlings showed markedly elevated brain concentrations and pronounced salivation, tremors, choreoathetosis, and eventual fatalities. Plasma DLT levels did not reliably reflect brain levels over time. Plasma:brain ratios were time and dose dependent, but apparently not age dependent. Brain levels were better correlated with the magnitude of salivation and tremors than plasma levels. Hepatic intrinsic clearance of DLT progressively increased during maturation, as did the hepatic extraction ratio. Thus, limited capacity to metabolically inactivate DLT appeared primarily responsible for the inordinately high target organ doses and acute neurotoxicity in pups and weanling rats. Hepatic blood flow was not rate limiting in any age group. Limited DLT hydrolysis was manifest in vivo in the pups by relatively low plasma PBA levels. Elevated exposure of the immature brain to a pyrethroid may prove to be of consequence for long-term, as well as short-term neurotoxicity.

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.

Choreathetosis due to abuse of levothyroxine.

Choreathetosis due to thyrotoxicosis is a rare movement disorder of acute onset. Here we present the first report of choreathetosis secondary to abuse of levothyroxine. A 35-year-old woman with autoimmune thyroiditis tripled her daily levothyroxine intake and lost 20 kg of weight during the following 6 months. She soon developed incapacitating choreathetosis. When levothyroxine was reduced to her usual dosage, all symptoms vanished in 7 days. The prompt effect of dose correction points towards a direct influence of levothyroxine on the basal ganglia; alternatively, the effects of levothyroxine might have been indirect and, possibly, autoimmune-mediated. Abuse of levothyroxine and related thyroid-tropic substances should be included into the differential diagnosis of acute choreathetosis.

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.

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.

Transient athetosis induced by tiagabine.

Movement disorders have been reported with use of different antiepileptic drugs (AEDs). We report a 32-year-old woman, affected by a symptomatic focal drug-resistant epilepsy and a mild hemiparesis, with acute athetoid movements, transiently linked to increasing tiagabine (TGB) therapy. To our knowledge, no other cases of acute athetosis related to TGB have been reported to date. However, we cannot rule out the possibility that involuntary movements were induced by an interaction between TGB and concomitant AEDs, in particular phenobarbital (PB), possibly by increasing GABAergic transmission. We hypothesize that the presence of a static encephalopathy may have influenced the kind of extrapyramidal side effect induced by TGB in our patient, leading to athetosis.