Intranasal disulfiram-induced encephalopathy: clinical and neuroimage findings
*Corresponding author: Dr. Ana Matas García. Hospital Clínic. Carrer de Villarroel, 170. E-08036 Barcelona.
E-mail: anmatas@clinic.cat
Introduction: Disulfiram-induced-encephalopathy is a rare complication that has been well described in adults. Although it usually occurs in acute intoxication with high doses of disulfiram, late onset encephalopathy has also been reported. Some authors propose the inhibition of dopamine beta-hydroxylase mediated by toxic metabolites of disulfiram as the main responsible, but the exact mechanism remains unclear. The aim of this report was to describe the clinical and neuroimaging findings in an unusual case of acute encephalitis due to disulfiram toxicity associated to chronic intranasal consume.
Case report: A chronic alcoholic who referred snorted use of a very high dose of disulfiram without simultaneous alcohol intake developed an acute encephalopathy with a rapidly progressive respiratory failure. A characteristic neuroimage finding consisting in extensive bilateral symmetric involvement of both pallidal nuclei was described. Recovery and neurologic improvement were slow. Two months after the intoxication, the patient still had slight intentional tremor and a scheduled magnetic resonance imaging. showed evolution of symmetrical areas of cytotoxic edema to necrosis.
Conclusion: Disulfiram-induced neurotoxicity must be suspect during chronic therapy with disulfiram or after acute ingestion of high doses. Symptoms such as symmetric sensory and motor neuropathy, confusion, catatonia, parkinsonism, ataxia, choreoathetosis, seizures and encephalopathy should make us rule out this disorder. A brain imaging test should be performed in these patients since a characteristic involvement of both nuclei pallidus has been described, but it is not present in all patients.
Caso clínico Paciente de 48 años con enolismo crónico que refirió el uso inhalado por vía intranasal de una dosis muy elevada de disulfiram sin ingesta simultánea de alcohol desarrolló una encefalopatía aguda con insuficiencia respiratoria rápidamente progresiva. La neuroimagen reveló una extensa afectación simétrica bilateral de ambos núcleos pálidos, un hallazgo característico en esta intoxicación. La recuperación neurológica fue lenta. Dos meses después de la intoxicación, el paciente presentaba un ligero temblor intencional residual y una resonancia magnética mostró una evolución de las áreas simétricas de edema citotóxico a necrosis.
Conclusión La neurotoxicidad inducida por disulfiram debe sospecharse durante el tratamiento crónico con disulfiram o después de una ingesta aguda de dosis elevadas. La presencia de síntomas como una neuropatía sensitivomotora simétrica, confusión, catatonía, parkinsonismo, ataxia, coreoatetosis, convulsiones y encefalopatía nos debe obligar a descartar este trastorno. La neuroimagen debe considerarse en este escenario, ya que se ha descrito una afectación característica de ambos núcleos pálidos.