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Infant with right hemiplegia due to acute encephalopathy with biphasic seizures and late reduced diffusion (AESD): A
Ai Takahashi1,2, Erina Kamei1, Yuri Sato1
1Division of Rehabilitation Medicine, University of Fukui Hospital.
Insights
Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is rare, but rehabilitation helps motor recovery. Early intervention is key for children with AESD-related hemiplegia and developmental delays.
Area of Science:
- Pediatric neurology
- Infectious diseases
- Neurorehabilitation
Background:
- Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is a severe neurological condition often linked to infections in children.
- AESD typically presents in early childhood with characteristic MRI findings ('bright tree appearance') and can lead to significant neurological deficits.
- Hemiplegia is an uncommon sequela of AESD, with limited case reports available.
Observation:
- A case study of an 11-month-old girl diagnosed with AESD and hemiconvulsion-hemiplegia-epilepsy (HHE) syndrome.
- The patient presented with right-sided hemiplegia.
- Human herpesvirus 6 was identified as the causative agent through DNA testing.
Findings:
- The patient received pharmacotherapy and intensive rehabilitation therapy.
- Significant recovery in gross motor function was observed.
- Mild developmental delay was noted at 30 months of age.
Implications:
- This case highlights the rarity of AESD-induced hemiplegia and the effectiveness of rehabilitation in improving physical function.
- Despite functional recovery, ongoing monitoring for developmental delays is crucial for children with AESD.
- Early referral to specialized facilities is recommended for children with AESD to ensure comprehensive support for long-term development and schooling.
Rationale:
Acute 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 Concerns:
A case with right-sided hemiplegia due to AESD in an 11-month-old girl who was followed up to 30 mo of age.
Diagnoses:
The 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.
Interventions:
Pharmacotherapy and rehabilitation therapy.
Outcome:
Gross motor function has recovered considerably, but she had a mild developmental delay at 30 mo old.
Lessons:
Hemiplegia 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.
