Case Report
Copyright ©The Author(s) 2023. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Immunol. May 31, 2023; 13(1): 1-10
Published online May 31, 2023. doi: 10.5411/wji.v13.i1.1
Challenges to associate early onset epilepsy with COVID-19 autoimmune encephalitis: A case report
Salma Zouari Mallouli, Olfa Jallouli, Wafa Bouchaala, Sihem Ben Nsir, Fatma Kamoun Feki, Chahnez Charfi Triki
Salma Zouari Mallouli, Olfa Jallouli, Wafa Bouchaala, Sihem Ben Nsir, Fatma Kamoun Feki, Chahnez Charfi Triki, Department of Child Neurology, Hedi Chaker Sfax University Hospital and Research Laboratory LR19ES15-University of Sfax, Tunisia, Sfax 3029, Tunisia
Author contributions: Zouari Mallouli S contributed to manuscript writing and editing, and bibliography; Jallouli O, Bouchaala W, Ben Nsir S contributed to bibliography; Kamoun Feki F, Charfi Triki C contributed to editing and supervision; all authors have read and approved the final manuscript.
Informed consent statement: The patients' legal guardian provided informed written consent prior to study enrollment.
Conflict-of-interest statement: All the authors declare that they have no conflict of interest to disclose.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Salma Zouari Mallouli, Doctor, Medical Assistant, Department of Child Neurology, Hedi Chaker Sfax University Hospital and Research Laboratory LR19ES15-University of Sfax, El Ain Street Km 0.5, 3029 Sfax, Tunisia. mallouli.salma26@gmail.com
Received: December 11, 2022
Peer-review started: December 11, 2022
First decision: January 3, 2023
Revised: January 14, 2023
Accepted: February 2, 2023
Article in press: February 2, 2023
Published online: May 31, 2023
Processing time: 169 Days and 14.7 Hours
Abstract
BACKGROUND

Diagnosis of coronavirus disease 2019 (COVID-19)-related neurological events in the pediatric population is challenging. Overlapping clinical picture of children with altered neurological state and inborn errors of metabolism, in addition to the frequency of asymptomatic COVID-19 cases, pose the main challenges for diagnosis. Diagnostic approaches to the onset post-COVID 19 subacute encephalopathy are still troublesome as seronegative autoimmune encephalitis (AIE) is reported.

CASE SUMMARY

A 27-mo-old boy was admitted for stormy refractory seizure of polymorphic semiology and altered mental status followed by various neuropsychiatric features that were suggestive of AIE. Brain magnetic resonance imaging and cerebrospinal fluid analysis were normal. Neither the immunological assessment, including viral serologies, antinuclear antibodies, autoimmune antibodies (NMDA, AMPA, CASPR2, LG11, GABARB, Hu, Yo, Ri, CV2, PNMA2, SOX1, Titin, amphiphysin, Recoverin), nor the metabolic assessment for lactate and pyruvate showed significant anomaly. Both positive history of COVID-19 infection and the findings of characteristic repetitive extreme delta brush played a key role in the diagnosis of COVID-19-related AIE. A remarkable improvement in the state of the child was noted after two pulse doses of intravenous Veino-globulin and high dose of intravenous Corticosteroid.

CONCLUSION

Diagnostic biomarkers for AIE might aid effective treatment.

Keywords: Post-COVID-19; Auto-immune encephalitis; Diagnosis biomarkers; Pediatric population; Delta brush sign; Autoimmune antibodies

Core Tip: Overlapping epileptic syndromes and etiologies in infancy constitute a real challenge to the diagnostic and therapeutic approach to autoimmune epilepsy related to post-coronavirus disease 2019 (COVID-19) autoimmune encephalitis (AIE). Indeed, epileptic seizures could entail the onset of seronegative form of post-COVID-19 AIE. Adequate seizure management and control might be hampered by potential misdiagnosis. Defining clinical and paraclinical biomarkers of seronegative form of post-COVID-19 AIE would enable early diagnosis and management, and better prognosis.