Observational Study
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Psychiatry. Jul 19, 2022; 12(7): 958-969
Published online Jul 19, 2022. doi: 10.5498/wjp.v12.i7.958
Clinical characteristics of pediatric patients with treatment-refractory Tourette syndrome: An evidence-based survey in a Chinese population
Ying Li, Jun-Juan Yan, Yong-Hua Cui
Ying Li, Jun-Juan Yan, Yong-Hua Cui, Department of Psychiatry, Beijing Children's Hospital, Beijing 100045, China
Author contributions: Li Y and Yan JJ contribute equally to this study; Cui YH and Li Y took the initiative; Yan JJ participated in the data collection; Li Y performed the data analysis; Yan JJ finished the draft; all authors have read and approved the manuscript.
Supported by the National Natural Science Foundation of China (NSFC), No. 82171538; and the Beijing Natural Science Foundation, No. 7212035.
Institutional review board statement: The study was reviewed and approved by the Institutional Review Board of the Beijing Children’s Hospital (No. 2021-82171538).
Conflict-of-interest statement: All other authors report no conflict of interest for this article.
Data sharing statement: Data is available upon reasonable request for clearly defined scientific purposes from the corresponding author at cuiyonghua@bch.com.cn.
STROBE statement: The authors have read the STROBE Statement checklist of items, and the manuscript was prepared and revised according to the STROBE Statement checklist of items.
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: Yong-Hua Cui, MD, Chief Doctor, Department of Psychiatry, Beijing Children's Hospital, No. 56 Nanlishi Road, Beijing 100045, China. cuiyonghua@bch.com.cn
Received: December 21, 2021
Peer-review started: December 21, 2021
First decision: March 13, 2022
Revised: April 6, 2022
Accepted: June 27, 2022
Article in press: June 27, 2022
Published online: July 19, 2022
Processing time: 210 Days and 2.7 Hours
Abstract
BACKGROUND

Tourette syndrome (TS) is a complex neurodevelopmental condition marked by tics, as well as a variety of psychiatric comorbidities, such as obsessive-compulsive disorders (OCDs), attention deficit hyperactivity disorder (ADHD), anxiety, and self-injurious behavior. TS might progress to treatment-refractory Tourette syndrome (TRTS) in some patients. However, there is no confirmed evidence in pediatric patients with TRTS.

AIM

To investigate the clinical characteristics of TRTS in a Chinese pediatric sample.

METHODS

A total of 126 pediatric patients aged 6-12 years with TS were identified, including 64 TRTS and 62 non-TRTS patients. The Yale Global Tic Severity Scale (YGTSS), Premonitory Urge for Tics Scale (PUTS), and Child Behavior Checklist (CBCL) were used to assess these two groups and compared the difference between the TRTS and non-TRTS patients.

RESULTS

When compared with the non-TRTS group, we found that the age of onset for TRTS was younger (P < 0.001), and the duration of illness was longer (P < 0.001). TRTS was more often caused by psychosocial (P < 0.001) than physiological factors, and coprolalia and inappropriate parenting style were more often present in the TRTS group (P < 0.001). The TRTS group showed a higher level of premonitory urge (P < 0.001), a lower intelligence quotient (IQ) (P < 0.001), and a higher percentage of family history of TS. The TRTS patients demonstrated more problems (P < 0.01) in the “Uncommunicative”, “Obsessive-Compulsive”, “Social-Withdrawal”, “Hyperactive”, “Aggressive”, and “Delinquent” subscales in the boys group, and “Social-Withdrawal” (P = 0.02) subscale in the girls group.

CONCLUSION

Pediatric TRTS might show an earlier age of onset age, longer duration of illness, lower IQ, higher premonitory urge, and higher comorbidities with ADHD-related symptoms and OCD-related symptoms. We need to pay more attention to the social communication deficits of TRTS.

Keywords: Treatment-refractory Tourette syndrome, Yale Global Tic Severity Scale, Child Behavior Checklist, Premonitory Urge for Tics Scale, Social withdrawal, Obsessive-compulsive disorder

Core Tip: This study provides important evidence of treatment-refractory Tourette syndrome (TS) among Chinese patients due to the current shortage of studies based on Chinese samples. We found that the onset age of pediatric patients with treatment-refractory TS (TRTS) might be younger, and they might have a longer duration of illness, a lower intelligence quotient, and a higher premonitory urge, which often fluctuate due to psychosocial factors. Moreover, TRTS children might suffer more emotional and behavioral problems including social communication deficits (such as uncommunicative and social withdrawal), attention deficit hyperactivity disorder-related symptoms (hyperactive, aggressive, and delinquent), and obsessive-compulsive symptoms. These were the basic clinical characteristics of TRTS based on Chinese pediatric patients. Unravelling these clinical characteristics is beneficial for the early diagnosis and treatment of TRTS.