Copyright
©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Oct 26, 2020; 8(20): 4866-4875
Published online Oct 26, 2020. doi: 10.12998/wjcc.v8.i20.4866
Published online Oct 26, 2020. doi: 10.12998/wjcc.v8.i20.4866
Unremitting diarrhoea in a girl diagnosed anti-N-methyl-D-aspartate-receptor encephalitis: A case report
Norrapat Onpoaree, Faculty of Medicine, Chulalongkorn University, Bangkok 10330, Thailand
Montida Veeravigrom, Division of Neurology, Department of Pediatrics, Faculty of Medicine, Chulalongkorn University, Bangkok 10330, Thailand
Anapat Sanpavat, Division of Pathology, Faculty of Medicine, Chulalongkorn University, Bangkok 10330, Thailand
Narissara Suratannon, Division of Allergy, Department of Pediatrics, Faculty of Medicine, Chulalongkorn University, Bangkok 10330, Thailand
Palittiya Sintusek, Division of Gastroenterology and Pediatric Gastroenterology and Hepatology Research Unit, Department of Pediatrics, Faculty of Medicine, Chulalongkorn University, Bangkok 10330, Thailand
Author contributions: Onpoaree N and Sintusek P reviewed the literature and drafted the manuscript; Veeravigrom M contributed to manuscript draft, interpreted the electroencephalography and made clinical correlation with brain images; Sunpavat A interpreted the histopathological data and provided the histopathology images; Surathannon N interpreted the immunologic study; Sintusek P performed endoscopy, provided the endoscopic images; and all authors approved the final manuscript.
Supported by The Pediatric Gastroenterology and Hepatology Research Unit , Thailand; Grants for Development of New Faculty Staff , Ratchadaphiseksomphot Endowment Fund, Thailand, No. RA63/012; and Thailand Research Fund Thailand Science Research and Innovation , Thailand, No. MRG6280190.
Informed consent statement: Informed written consent was obtained from the patient's guardian (mother) for publication of this report and any accompanying images.
Conflict-of-interest statement: All authors declare that they have no conflict of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist, 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: Palittiya Sintusek, MD, MSc, Assistant Professor, Lecturer, Division of Gastroenterology and Pediatric Gastroenterology and Hepatology Research Unit, Department of Pediatrics, Faculty of Medicine, Chulalongkorn University, 1873, Rama 4 Road, Pathumwan, Bangkok 10330, Thailand. palittiya.s@chula.ac.th
Received: April 11, 2020
Peer-review started: April 11, 2020
First decision: June 13, 2020
Revised: July 27, 2020
Accepted: September 25, 2020
Article in press: September 25, 2020
Published online: October 26, 2020
Processing time: 194 Days and 4 Hours
Peer-review started: April 11, 2020
First decision: June 13, 2020
Revised: July 27, 2020
Accepted: September 25, 2020
Article in press: September 25, 2020
Published online: October 26, 2020
Processing time: 194 Days and 4 Hours
Core Tip
Core Tip: We report a girl with behavioural change who was finally diagnosed with anti-N-methyl-D-aspartate-receptor (anti-NMDAR) encephalitis. She had unremitting vomiting and diarrhoea after 43 d of corticosteroid therapy and was later diagnosed with cytomegalovirus (CMV) esophago-enterocolitis. Surprisingly, these symptoms subsided after antiviral therapy followed by repeated pulse methylprednisolone targeting the anti-NMDAR encephalitis. CMV esophago-enterocolitis in a patient with anti-NMDAR encephalitis has not been reported before and is very rare in patients who receive only moderate-dose corticosteroids. We highlighted that immune dysregulation in autoimmune encephalitis and the linkage of anti-NMDAR receptors and the enteric nervous system might be the cause of severe gastrointestinal involvement.