Adrenocorticotropic Hormone (ACTH)-Induced Dyskinesias in Infantile Spasms: A Video Case Report

Abstract

Case series Patients: Male, 10-month-old • Male, 8-month-old • Male, 5-month-old • Female, 4-month-old Final Diagnosis: ACTH-induced dyskinesias Symptoms: ACTH-induced dyskinesias Medication: — Clinical Procedure: — Specialty: Neurology Objective: Rare disease Background: Infantile spasms is an age-specific epilepsy syndrome that occurs during infancy and is characterized by tonic and/or flexor-extensor spasms, hypsarrhythmia on electroencephalography (EEG), and neurodevelopmental regression. Adrenocorticotropic hormone (ACTH) is considered one of the main therapies for the treatment of infantile spasms, but despite its great efficacy, it is still associated with potential adverse effects. Case Reports: Four patients previously diagnosed with infantile spasms were treated with ACTH following the usual treatment regimen. All patients developed asymmetric, involuntary movements, with phenomenology characteristic of dyskinesia. The patients did not manifest loss of consciousness, and the EEG did not show epileptic activity during those episodes. In all cases, involuntary movements disappeared after the completion of the hormonal therapy. Conclusions: The adverse effect of hormonal therapy in infantile spasms is not well known in the literature and could be mistaken as seizures, leading to inappropriate management.

Adrenocorticotropic Hormone-Induced Dyskinesia and Probable Sudden Unexpected Death in an Infant with Early Infantile Developmental and Epileptic Encephalopathy—Infantile Epileptic Spasm Syndrome Overlap

“…In his series too, facial dyskinesias were the most common, apart from asymmetric limb movements and uprolling of eyeballs which was interrupted by touch. 22 The dyskinesias were partly responsive to diphenhydramine and biperiden, and the treatment courses were completed in all cases, unlike ours, where progressive choreiform movements forced us to stop ACTH. Arita et al 23 described ACTH-induced dyskinesias (arm elevation and tongue protrusion) on the 7th day of treatment and subsiding 2 months after ACTH discontinuation.…”

“…The earliest onset was after 2 weeks of treatment initiation and resolution was after 1 month of treatment cessation. Crippa et al 22 described 4 cases of ACTH-induced dyskinesias, onset varying from 4 to 10 days after starting treatment and resolution occurring 1 to 4 weeks after stopping ACTH. In his series too, facial dyskinesias were the most common, apart from asymmetric limb movements and uprolling of eyeballs which was interrupted by touch.…”