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
ARTICLE HIGHLIGHTS
Research 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. However, no Chinese version of the TRTS criteria has been described. Moreover, the different criteria for TRTS were established mostly based on the clinical characteristics of adult patients with Tourette syndrome.

Research motivation

We need more confirmatory evidence about the clinical characteristics of TRTS. However, few studies have focused on the behavioral and emotional components of TRTS. Identifying the “indicators” of TRTS in the early stage may help in the treatment of these patients. Whether TRTS is different from “pure TS” (only tic symptoms without comorbidities) is unknown. More evidence is needed to explore these differences, especially at the early stage of TRTS.

Research objectives

This study aimed to examine the clinical characteristics of TRTS in a Chinese pediatric population, compare the clinical characteristics (i.e., the onset of tic age, duration of illness, intelligence quotient (IQ), and behavioral and emotional problems) of patients with TRTS and non-TRTS patients, and report the locations and the frequency of tic onset in TRTS.

Research 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 groups. Descriptive statistics were performed to identify the basic clinical information, and t tests or χ2 tests were used to compare the different variables of different TS groups.

Research 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.

Research conclusions

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 than ‘pure TS’. Moreover, TRTS shows more social communication deficits that need to be covered in both the assessment and treatment of TRTS. TRTS might be one of the subtypes of TS. We need to develop a proper Chinese version definition of the TRTS in the future, especially for pediatric patients.

Research perspectives

In previous studies, we focused more on ADHD and obsessive-compulsive symptoms in TRTS, and social communication-related problems seemed to be neglected. It should be noted that social communication deficits are crucial signs of functional impairment, suggesting that we also need to assess social communication deficits during the assessment of function in TRTS. Therefore, we should pay more attention to social communication deficits in TRTS regardless of the assessment or the treatment.