Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Aug 16, 2021; 9(23): 6916-6921
Published online Aug 16, 2021. doi: 10.12998/wjcc.v9.i23.6916
Dopamine agonist responsive burning mouth syndrome: Report of eight cases
Qi-Cui Du, Ying-Ying Ge, Wen-Lin Xiao, Wei-Fei Wang
Qi-Cui Du, Department of Stomatology, Liaocheng People's Hospital, Liaocheng 252000, Shandong Province, China
Ying-Ying Ge, School of Stomatology, Qingdao University, Qingdao 266003, Shandong Province, China
Wen-Lin Xiao, Department of Stomatology, Affiliated Hospital of Qingdao University, Qingdao 266003, Shandong Province, China
Wei-Fei Wang, Department of Neurology, Liaocheng People's Hospital, Liaocheng 252000, Shandong Province, China
Author contributions: Du QC and Wang WF were responsible for the data collection; Du QC and Ge YY drafted the manuscript and interpreted the results; Wang WF and Xiao WL designed the study and were the project supervisors; all authors reviewed and approved the final manuscript.
Informed consent statement: Written informed consent was obtained from the patients for the publication of this report and the accompanying images.
Conflict-of-interest statement: The authors declare no conflicts of interest for this 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: Wei-Fei Wang, MA, Chief Doctor, Department of Neurology, Liaocheng People's Hospital, No. 67 Dongchangxi Road, Liaocheng 252000, Shandong Province, China. wenlinxiaocn@163.com
Received: April 22, 2021
Peer-review started: April 22, 2021
First decision: May 24, 2021
Revised: June 4, 2021
Accepted: June 15, 2021
Article in press: June 15, 2021
Published online: August 16, 2021
Processing time: 105 Days and 6.5 Hours
Abstract
BACKGROUND

Burning mouth syndrome (BMS) is characterized by burning sensation of the oral mucosa. There is a lack of effective treatment. In recent years, a special subtype of BMS has been reported, in which oral burning sensation is alleviated after chewing, speaking, or dopaminergic drug delivery. Currently, there are few reports about the subtype of BMS in China. This study was a retrospective analysis of the clinical data of BMS patients sensitive to dopamine agonist at our hospital, aiming to improve the recognition on this disease.

CASE SUMMARY

Eight patients diagnosed with dopamine agonist responsive BMS at the Liaocheng People's Hospital from January 1, 2017 to June 30, 2020 were recruited. The clinical manifestations, treatment, and prognosis were retrospectively analyzed. There were three male and five females in the eight patients. The median age was 56 years (range, 46-65 years). All the eight patients showed burning pain in the mouth. The symptoms were mild in the morning and severe in the evening, and alleviated after chewing, talking, and other oral activities. Four patients were accompanied by restless legs syndrome (RLS). Family history of RLS was positive in two patients. All patients were treated with pramipexol, and symptoms were basically relieved after 2-8 wk.

CONCLUSION

Dopamine agonist responsive BMS is a special subtype of BMS, which is alleviated after oral activities. Dopamine receptor agonist is an effective treatment.

Keywords: Burning mouth syndrome; Restless legs syndrome; Dopamine receptor agonists; Chinese; Case report

Core Tip: Dopamine agonist responsive burning mouth syndrome (BMS) is a special subtype of BMS. We retrospectively summarized the clinical data of eight patients with a diagnosis of dopamine agonist responsive BMS in China to improve the recognition on this disease. All patients showed burning pain in the mouth, which was mild in the morning and severe in the evening, and alleviated after chewing, talking, and other oral activities. Four patients were accompanied by restless legs syndrome (RLS). Family history of RLS was positive in two patients. All patients were treated with pramipexol, and symptoms were basically relieved after 2-8 wk.