SARS-CoV-2-Related Pediatric Encephalopathy with Biphasic Convulsions and Cortical Blindness: A Case Report

Abstract

Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is an encephalopathy syndrome that develops during the febrile phase of infection and is characterized by biphasic seizures and bright tree appearance (BTA) on imaging. In this study, we examined a 1-year-old child who was diagnosed with AESD caused by severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection. He had a fever for 2 days and was diagnosed with coronavirus disease 2019 on the day of consultation based on a positive antigen test for SARS-CoV-2. The seizures had stopped on arrival to hospital; however, symptoms of encephalopathy persisted. Despite being treated for acute encephalopathy with steroid pulse therapy, vitamins, and remdesivir, he had poor expressive language, and electroencephalography showed generalized slow waves. A repeat head magnetic resonance imaging showed BTA in the parietal and occipital lobes, and seizures recurred during hospital stay. He was also found to have cortical blindness. At the 4-month follow-up, the vision gradually improved and the hand movements recovered. SARS-CoV-2 infection can cause febrile convulsions, which can lead to the development of AESD. Furthermore, a subtype of AESD has occipital lobe predominant lesions that can cause visual impairment.

Keywords

acute encephalopathy - biphasic seizures - reduced diffusion - bright tree appearance

Ethics Approval and Consent to Participate

Informed consent was obtained from the patient's parents for the publication of this report. The Independent Ethics Committee approved this study (R4–156).

Publication History

Received: 29 January 2023

Accepted: 20 March 2023

Article published online:
13 April 2023

• References

• 1 Ito Y, Natsume J, Kidokoro H. et al. Seizure characteristics of epilepsy in childhood after acute encephalopathy with biphasic seizures and late reduced diffusion. Epilepsia 2015; 56 (08) 1286-1293
• 2 Hoshino A, Saitoh M, Oka A. et al. Epidemiology of acute encephalopathy in Japan, with emphasis on the association of viruses and syndromes. Brain Dev 2012; 34 (05) 337-343
• 3 Kasai A, Shimizu J, Sato M, Kitamura M, Inaba Y, Motobayashi M. Reduced impact of viral load of HHV-6 in liquor on severity of AESD due to exanthema subitum: a case report and literature review. Brain Dev 2021; 43 (08) 879-883
• 4 Shoji K, Akiyama T, Tsuzuki S. et al. Clinical characteristics of COVID-19 in hospitalized children during the Omicron variant predominant period. J Infect Chemother 2022; 28 (11) 1531-1535
• 5 Tada H, Takanashi J, Okuno H. et al. Predictive score for early diagnosis of acute encephalopathy with biphasic seizures and late reduced diffusion (AESD). J Neurol Sci 2015; 358 (1-2): 62-65
• 6 Yokochi T, Takeuchi T, Mukai J. et al. Prediction of acute encephalopathy with biphasic seizures and late reduced diffusion in patients with febrile status epilepticus. Brain Dev 2016; 38 (02) 217-224
• 7 Mizuguchi M, Ichiyama T, Imataka G. et al. Guidelines for the diagnosis and treatment of acute encephalopathy in childhood. Brain Dev 2021; 43 (01) 2-31
• 8 Fukui KO, Kubota M, Terashima H, Ishiguro A, Kashii H. Early administration of vitamins B1 and B6 and l-carnitine prevents a second attack of acute encephalopathy with biphasic seizures and late reduced diffusion: a case control study. Brain Dev 2019; 41 (07) 618-624
• 9 Panda PK, Sharawat IK, Panda P, Natarajan V, Bhakat R, Dawman L. Neurological complications of SARS-CoV-2 infection in children: a systematic review and meta-analysis. J Trop Pediatr 2021; 67 (03) fmaa070
• 10 Morino S, Goto T, Ohtaki U, Miyama S. [Acute encephalopathy with biphasic seizures and late reduced diffusion with visual disturbance and higher brain dysfunction]. No To Hattatsu 2011; 43 (04) 295-299
• 11 Randhawa MS, Randhawa TS, Angurana SK, Ratho RK. Acute encephalopathy with biphasic seizures and late restricted diffusion temporally associated with human bocavirus infection. BMJ Case Rep 2022; 15 (07) e251019
• 12 Takanashi J, Oba H, Barkovich AJ. et al. Diffusion MRI abnormalities after prolonged febrile seizures with encephalopathy. Neurology 2006; 66 (09) 1304-1309 , discussion 1291
• 13 Chaudhry FB, Raza S, Ahmad U. Anton's syndrome: a rare and unusual form of blindness. BMJ Case Rep 2019; 12 (12) e228103
• 14 Melnick MD, Tadin D, Huxlin KR. Relearning to see in cortical blindness. Neuroscientist 2016; 22 (02) 199-212
• 15 Saitoh M, Shinohara M, Hoshino H. et al. Mutations of the SCN1A gene in acute encephalopathy. Epilepsia 2012; 53 (03) 558-564