Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jul 6, 2021; 9(19): 5325-5331
Published online Jul 6, 2021. doi: 10.12998/wjcc.v9.i19.5325
Glutamic acid decarboxylase 65-positive autoimmune encephalitis presenting with gelastic seizure, responsive to steroid: A case report
Camerdy Yue Yang, Sheng-Ta Tsai
Camerdy Yue Yang, Sheng-Ta Tsai, Department of Neurology, China Medical University Hospital, Taichung 404332, Taiwan
Author contributions: Yang CY was the consulting neurology resident who evaluated the patient and initiated the case report and reviewed the literature and drafted the manuscript; Tsai ST was the attending physician in charge of patient’s treatment plan and revised the manuscript; all authors issued final approval for the version to be submitted.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying image.
Conflict-of-interest statement: All the authors have no conflict of interest related to the manuscript.
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: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Sheng-Ta Tsai, MD, Attending Doctor, Department of Neurology, China Medical University Hospital, No. 2 Yude Road, North District, Taichung 404332, Taiwan. d27056@mail.cmuh.org.tw
Received: February 26, 2021
Peer-review started: February 26, 2021
First decision: March 27, 2021
Revised: April 2, 2021
Accepted: May 20, 2021
Article in press: May 20, 2021
Published online: July 6, 2021
Processing time: 117 Days and 8.1 Hours
Abstract
BACKGROUND

Anti-glutamic acid decarboxylase (GAD) antibody is known to cause several autoimmune-related situations. The most known relationship is that it may cause type I diabetes. In addition, it was also reported to result in several neurologic syndromes including stiff person syndrome, cerebellar ataxia, and autoimmune encephalitis. Decades ago, isolated epilepsy associated with anti-GAD antibody was first reported. Recently, the association between temporal lobe epilepsy and anti-GAD antibody has been discussed. Currently, with improvements in examination technique, many more autoimmune-related disorders can be diagnosed and treated easier than in the past.

CASE SUMMARY

A 44-year-old female Asian with a history of end-stage renal disease (without diabetes mellitus) under hemodialysis presented with diffuse abdominal pain. The initial diagnosis was peritonitis complicated with sepsis and paralytic ileus. Her peritonitis was treated and she recovered well, but seizure attack was noticed during hospitalization. The clinical impression was gelastic seizure with the presentation of frequent smiling, head turned to the right side, and eyes staring without focus; the duration was about 5–10 s. Temporal lobe epilepsy was recorded through electroencephalogram, and she was later diagnosed with anti-GAD65 antibody positive autoimmune encephalitis. Her seizure was treated initially with several anticonvulsants but with poor response. However, she showed excellent response to intravenous methylprednisolone pulse therapy. Her consciousness returned to normal, and no more seizures were recorded after 5 d of intravenous methylprednisolone treatment.

CONCLUSION

In any case presenting with new-onset epilepsy, in addition to performing routine brain imaging to exclude structural lesion and cerebrospinal fluid studies to exclude common etiologies of infection and inflammation, checking the autoimmune profile has to be considered. In the practice of modern medicine, autoimmune-related disorders are relatively treatable and should not be missed.

Keywords: Anti-GAD antibody; GAD65 antibody; Autoimmune encephalitis; Gelastic seizure; Electroencephalogram; Case report

Core Tip: This case reminds us that autoimmune encephalitis is a diagnosis that should not be missed when we encountering a patient presenting with new-onset seizure. Gelastic seizure could be a rare presentation of glutamic acid decarboxylase 65-positive autoimmune encephalitis.