Infantile spasm-like paroxysms are often difficult to classify as epileptic or non-epileptic. We aimed to study spontaneously relieved (non-epileptic) spasms of infancy in sleep. Elucidation of the electroclinical characteristics and differential diagnoses of such spasms could facilitate accurate diagnosis in the future.
We retrospectively analyzed the clinical data, video-EEG recordings, and other laboratory test results of patients with spontaneously relieved spasms of infancy during sleep. All the enrolled patients were followed up for at least 5 months.
Seven infants were included in this study. The median age at spasm onset was 0.5 months (range: 0.1–2 months). The episodes were characterized by clusters of non-epileptic spasms of the head, trunk, or extremities lasting approximately 0.5–2 s, and were validated by ictal electromyography (EMG)/video EEG. Episodes occurred several times daily in clusters, particularly during sleep; two patients also experienced episodes while awake. Additionally, non-epileptic jerks were recorded in 3 patients. All non-epileptic spasms were completely resolved 2 weeks to 3 months after onset. Moreover, neuropsychomotor development in all patients was normal at the last follow-up (5 to 12 months).
Spontaneously relieved spasms of infancy in sleep is a self-limiting movement disorder characterized by onset between 0.1 and 2 months of age, and by clusters of spasms occurring in sleep. Correct differential diagnosis relies on familiar clinical and electrophysiological features.
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Published online: June 16, 2022
Accepted: June 3, 2022
Received in revised form: May 12, 2022
Received: December 9, 2021
© 2022 The Japanese Society of Child Neurology Published by Elsevier B.V. All rights reserved.