• Ai Takahashi, Erina Kamei, Yuri Sato, Seiichiro Shimada, Misao Tsubokawa, Genrei Ohta, Yusei Ohshima, and Akihiko Matsumine.
• Division of Rehabilitation Medicine, University of Fukui Hospital.
• Medicine (Baltimore). 2021 Jun 4; 100 (22): e25468e25468.

RationaleAcute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is a condition characterized by biphasic convulsions and disturbance of consciousness. In Japan, the most common pediatric cases of acute encephalopathy are associated with infection. AESD usually occurs in early childhood, with the characteristic magnetic resonance imaging (MRI) appearance called "bright tree appearance." The disease often has neurological sequelae and interferes with the schooling of children and their activities of daily living; however, there are few clinical case reports of hemiplegia caused by AESD.Patient ConcernsA case with right-sided hemiplegia due to AESD in an 11-month-old girl who was followed up to 30 mo of age.DiagnosesThe patient was diagnosed with overlap AESD and hemiconvulsion-hemiplegia-epilepsy syndrome (HHE syndrome), based on the clinical course and imaging findings. DNA tests of her blood and cerebrospinal fluid revealed the presence of human herpesvirus 6.InterventionsPharmacotherapy and rehabilitation therapy.OutcomeGross motor function has recovered considerably, but she had a mild developmental delay at 30 mo old.LessonsHemiplegia due to AESD was extremely rare, and appropriate rehabilitation treatment resulted in recovery of physical function. However, as mild developmental delay was observed, the patient was referred to a specialized facility before entering school.Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.

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