518822
research-article2014
JADXXX10.1177/1087054713518822Journal of Attention DisordersLiu et al.
Article
The Subjective Quality of Life in Young People With Tourette Syndrome in China
Journal of Attention Disorders 1–7 © 2014 SAGE Publications Reprints and permissions: sagepub.com/journalsPermissions.nav DOI: 10.1177/1087054713518822 jad.sagepub.com
Shiguo Liu1, Lanlan Zheng1, Xueping Zheng1, Xinhua Zhang1, Mingji Yi1, and Xu Ma2,3
Abstract Objective: To explore the subjective quality of life (QoL) in children with Tourette Syndrome (TS) in China to provide a basis for more effective interference. Method: A total of 107 patients and 107 controls were enrolled. Subjective QoL was investigated by Inventory of Subjective Life Quality, Family Environment Scale of Chinese Version, and the Yale Global Tic Severity Scale, and a case–control study was performed. Results: The total score of subjective QoL and family life, school life, peer relationship, cognitive component, environment, self-awareness, cognitive component and depression experience in the TS were lower than control. Patients with co-morbid exhibited significantly lower scores within the subjective QoL family life, peer relationship, school life, self-awareness, and cognitive affective domains. Conclusion: The subjective QoL is impaired and it is important to control clinical symptoms and improve family environment for the improvement of the subjective QoL in TS. (J. of Att. Dis. XXXX; XX(X) 1-XX) Keywords Tourette syndrome, quality of life, child, China
Introduction Tourette Syndrome (TS) is a neurodevelopmental disorder characterized by motor and vocal tics. Motor tics are sudden, repetitive, stereotyped, nonrhythmic movements, while vocal or phonic tics represent involuntary sounds produced by moving air through the nose, mouth, or throat (Singer, 2005). Clinical diagnosis of TS is based on that multiple motor and one or more vocal tics are present for more than a year, and during which there was never a ticfree period of more than three consecutive months (Leckman, Bloch, King, & Scahill, 2006; Malone & Pandya, 2006). However, tics are commonly deteriorated when patients undergo emotional stimuli such as psychosocial stress, fatigue, or adversities, while they tend to ease in a state of concentrating, pleased, or asleep (O’Connor, Brisebois, Brault, Robillard, & Loiselle, 2003; Wood et al., 2003). Typically, tics usually start around 8 years of age, peak early during the second decade of life, and decline by the end of adolescence (Bloch et al., 2006; Coffey et al., 2004; Leckman et al., 1998; Pappert, Goetz, Louis, Blasucci, & Leurgans, 2003). Complete resolution occurs by age 18 in 50% of patients (Leckman, Bloch, Scahill, & King, 2006), and only 20% or fewer of children with TS continue to present a moderate level of impairment of global functioning by the age of 20. Clinic data showed that males are five times more likely to be affected than females, and the prevalence may approach 0.72% (Dozier, 2006).
Although the hallmark of TS is the presence of motor and vocal tics, the clinical spectrum also includes a variety of psychiatric co-morbidities. Approximately 90% of TS patients experience a psychiatric co-morbidity (Kurlan et al., 2002), and the most common form is the ADHD (Freeman, 1997; Robertson, 2006). Around 65% of children with TS are highly associated with ADHD and the clinical spectrum of the two neurodevelopmental disorders tends to overlap (Sukhodolsky et al., 2003). This co-occurrence of TS and ADHD can be associated with disruptive behaviors such as aggression, explosive behavior, low frustration tolerance, and noncompliance (Black & Mink, 2000; Kurlan et al., 2002; Snider et al., 2002). The obsessive-compulsive disorder (OCD) is another common co-morbidity for TS. Over half of TS patients also have prominent OCD. Kano and co-workers reported the association between TS and co-morbidities in Japan, in which TS patients’ co-concurrence with OCD showed significant higher frequency for coprophenomana, impulsiveness, school refusal, self-injurious behaviors, and 1
Qingdao University, China Peking Union Medical College, Beijing, China 3 National Research Institute for Family Planning, Beijing, China 2
Corresponding Author: Mingji Yi, Child Healthcare Department, The Affiliated Hospital of Medical College, Qingdao University, 16 Jiangsu Road, Qingdao 266003, China. Email: [email protected]
Downloaded from jad.sagepub.com at PENNSYLVANIA STATE UNIV on November 14, 2015
2
Journal of Attention Disorders
clumsiness than that of patients with TS only (Kano, Ohta, Nagai, & Scahill, 2010). They further demonstrated that tic symptoms and impairment during the worst period were significantly severer in TS patients with OCD than that of patients with TS only. Other co-morbidities may also be apparent such as phobias, anxiety, aggressive, oppositional defiant disorder, depression, and other mood disorders (Kano et al., 2010). As preliminary research showed that up to 88% of patients consider that having tics interferes their life, considerable attention has been devoted to the study of subjective quality of life (QoL) in those people (Erenberg, Cruse, & Rothner, 1987). Perceived subjective QoL can determine the subjective experience of living with a health condition, affect planning for the future, and potentially affect acceptance of and adherence to treatment. There is a need to consider the features of TS that have the most significant impact on young people’s subjective QoL, so that clinicians can manage these effectively. The first study of QoL of patients with TS demonstrated that patients with TS showed significantly worse QoL than a general population sample (Elstner, Selai, Trimble, & Obertson, 2001). It is also reported that the subjective QoL of young people with tics in the United States was lower than that of healthy controls with respect to emotional, social, and school functioning but comparable in terms of physical functioning. Studies also showed that tic severity was inversely related to social acceptableness. The purpose of the present study was to evaluate subjective QoL in a large sample of young patients with TS in China from specialized outpatient clinics. We also analyzed whether tic severity or co-morbid (OCD and/or ADHD) impair the subjective QoL of patients with TS.
Material and Method Participants Participants were recruited from Department of Child Health Care, Affiliated Hospital of Qingdao University Medical College from 2008 until 2011. Participants included 107 young people with TS (92 males, 15 females), average age is M = 10.1 ± 1.1 and 107 control (59 males, 48 females), average age is 9.9 ± 1.0. The diagnoses of TS and other clinical conditions were made in accordance with Diagnostic and Statistical Manual of Mental Disorders (4th ed., text rev.; DSM-IV-TR; American Psychiatric Association, 2000) criteria by a psychiatrist with extensive experience. The control group had no psychiatric disorders, which undergo the same diagnostic evaluations that the TS group participants did.
Measures Inventory of Subjective Life Quality (ISLQ). The ISLQ (Freeman et al., 2000) evaluate two components (cognitive as
Points and the affective component) and eight dimensions (family life, peer relationship, School life, living environment, self-knowledge, the experience of depression, the experience of anxiety and physical emotion). The composition, dimensions and total score higher, indicating higher levels of satisfaction. Family Environment Scale of Chinese Version (FES-CV). FES was prepared in 1981, 90 questions, all of them are true or false, and takes about 30 min to complete (Kurlan, 1992). The scale is divided into 10 subscales; respectively evaluate 10 different families, social and environmental characteristics. In many countries, the FES has been widely used to describe different types of family characteristics and the family situation in state of crisis, evaluate the home environment changes in family intervention, as well as the comparison of other aspects of family environment and family life. FES’s evaluation of household characteristics include cohesion, expressiveness, conflict, independence, achievement orientation, intellectual-cultural orientation, active-recreational orientation, moral-religious emphasis, organization, and control. FES need to be modified to use in China, this is because some of the concepts assessed in the scale is not suitable for Chinese families, the contents of several projects in the Chinese cultural environment seem very inappropriate (such as independence, moral-religious emphasis, and expressiveness). Therefore, we used FES-CV and excluded three items (independence, moral-religious emphasis, and expressiveness). The Yale Global Tic Severity Scale (YGTSS). The YGTSS is a reliable clinician-rated interview (Leckman et al., 1989). The clinician initially notes the presence of motor and phonic tics based on child and parent reports over the past week, as well as behavioral observations. Then, the clinician rates the severity of motor and phonic tics on five separate dimensions each: number, frequency, intensity, complexity, and interference. Five index scores are obtained: Total Motor Tic Score, Total Phonic Tic Score, Total Tic Score, Overall Impairment Rating, and Global Severity Score. According to Global Severity Score, the severities of tic symptoms are divided into three grades: mild (50 points; Chang et al., 2008).
Statistics Data were analyzed using SPSS software, PC version 17.0 for Windows. Quantitative data were expressed by the means ± standard deviations (M ± SD), and group means were compared by t test and ANOVA. The first part of analysis described the general information of TS children and control children; the second part of analysis compared the subjective QoL satisfaction scores given by patients with TS to control. The third part of analysis investigated whether patients with TS who had more severe tics or co-morbid
Downloaded from jad.sagepub.com at PENNSYLVANIA STATE UNIV on November 14, 2015
3
Liu et al. Table 1. General Condition of TS Children and Control Children. Task
Category
Control
TS
χ2
p
Sex
man Female 2,500-4,000 kg Other Enlarge core Core Other Medium Other Junior and below High school College and above Junior and below High school College and above First child Second child and more Breast milk only Breast milk mainly Other
59 48 96 27 34 63 10 99 8 63 20
92 15 110 16 18 73 16 65 42 39 35 33 57 20 30 77 30 65 12 30
24.498
.000
3.730
.065
7.043
.030
30.169
.000
11.159
.004
2.620
.270
0.357
.654
25.602
.000
Born weight Family Household income Father’s education Mother’s education Children parity Feeding situation (4 months)
exhibited greater differences than the control groups on the measures administered. The last part of analysis explores the influencing factors of the subjective QoL satisfaction in children with TS.
Results Study of General Information of TS Children and Control Children There are significant difference between two groups of children in the sex ratio, family structure, father’s educational background, birth weight, family income, and feeding situation (4 months; p < .05 or p < .01), and no significant difference between two groups of children in birth order and mother’s educational background (Table 1).
Subjective QoL for Control and TS The second part of the analysis compared the subjective QoL scores given by patients with TS to control. Patients with TS scored significantly lower than control for family life, Peer relationship, School life, Environment, Selfawareness, Experience depression, Cognitive component, and total (p < .05; Table 2).
Subjective QoL for Severity Scale of TS First, we split patients with TS into two groups: mild and moderate to severe. Patients with moderate to severe had
24 22 20 73 34 57 40 10
significantly lower scores than milder TS for family life and cognitive component (p < 0.05). The second was dependent on the presence of co-morbidities, which generated five subgroups. The subgroups were independently compared with each other for scores on the scales administered. Subjective QoL may be more affected children with TS who suffer with other co-morbid conditions such as OCD and ADHD. The group of TS was split into four subgroups based on the presence of co-morbid conditions, so that they could be separately compared with controls. The subgroups were as follows: TS only (n = 52); TS plus OCD (TS + OCD: n = 9); TS plus ADHD (TS + ADHD: n = 34); TS plus OCD and ADHD (TS + OCD + ADHD: n = 12; Table 3). Patients with TS + OCD + ADHD exhibited significantly lower total subjective QoL score than control and significantly lower scores within the subjective QoL family life, peer relationship, school life, self-awareness, cognitive affective domains. These findings indicate that young people with TS + OCD + ADHD may experience social difficulties because of their symptoms and are perhaps less likely to spend time socializing with adults in the family at home.
Subjective QoL Factor Correlation Analysis Data from patients with TS were collected to investigate the correlation between factors and subjective QoL satisfaction score. The factors contain three feathers of Family Environment Scale, the age of patients, behavior score, and
Downloaded from jad.sagepub.com at PENNSYLVANIA STATE UNIV on November 14, 2015
4
Journal of Attention Disorders
Table 2. Mean and SD for Statistically Different Scores on the Subjective QoL for Control and TS. Measure Family life Peer relationship School life Environment Self-awareness Experience depression Anxiety experience The body emotion Cognitive component Affective component Total
Control (n = 107)
TS (n = 107)
t
p
7.19 ± 0.892 6.72 ± 1.559 6.31 ± 0.915 6.71 ± 1.402 6.39 ± 1.155 6.19 ± 1.894 5.63 ± 1.794 5.48 ± 2.2.107 68.69 ± 11.997 57.05 ± 18.281 65.97 ± 14.070
6.72 ± 1.026 6.07 ± 1.592 5.38 ± 1.146 6.14 ± 2.238 5.81 ± 1.573 5.47 ± 2.466 5.91 ± 2.196 4.87 ± 2.128 59.52 ± 11.942 56.05 ± 21.185 57.32 ± 15.447
3.556 3.037 6.524 2.891 3.071 2.394 −1.023 2.098 5.603 0.367 4.284
.000 .003 .000 .004 .002 .018 .308 .607 .000 .714 .000
Table 3. Mean and SD for Statistically Different Scores on the Subjective QoL for Control and the Co-Morbidity Subgroups. Measure Family life Peer relationship School life Environment Self-awareness Depression Anxiety Body emotion Cognitive Affective Total
Control (n = 107) 7.19 ± 0.892 6.72 ± 1.559 6.31 ± 0.915 6.21 ± 2.227 6.39 ± 1.155 6.19 ± 1.894 5.63 ± 1.794 5.48 ± 2.107 68.69 ± 11.997 57.05 ± 18.281 65.97 ± 14.070
TS only (n = 52) 7.17 ± 1.150 6.50 ± 1.639 5.38 ± 1.191a 6.38 ± 1.430 6.37 ± 1.442 5.85 ± 2.461 6.23 ± 2.454 4.94 ± 2.492 63.92 ± 12.811a 58.44 ± 26.312 62.10 ± 17.882
TS + OCD (n = 9)
TS + ADHD (n = 34) a,b
6.33 ± 1.000 5.89 ± 1.764 5.89 ± 0.928 5.78 ± 1.302 7.22 ± 1.202 6.56 ± 1.333 7.56 ± 1.333 4.67 ± 1.323 62.78 ± 9.770 67.89 ± 10.043 65.22 ± 6.360
TS + OCD + ADHD (n = 12)
a,b
6.47 ± 0.507 5.82 ± 1.507a,b 5.38 ± 1.231a 6.09 ± 1.240 4.97 ± 0.937a,b,c 5.38 ± 2.104 5.29 ± 1.697b,c 4.59 ± 1.811a 55.91 ± 7.921a,b 50.29 ± 13.543c 52.71 ± 9.114a,b,c
a,b,d
5.75 ± 0.452 5.00 ± 0.739a,b 5.00 ± 0.739a 5.50 ± 1.732 4.75 ± 2.006a,b,c 3.25 ± 2.989a,b,c,d 5.00 ± 1.954c 5.50 ± 1.732 48.25 ± 8.750a,b,c,d 43.75 ± 23.913a,b,c 43.75 ± 10.610a,b,c
F
p
11.377 5.075 11.628 0.685 14.054 5.794 3.709 1.543 14.482 2.870 10.915
.000 .001 .000 .603 .000 .000 .006 .191 .000 .024 .000
a
Compared with control, p < .05. Compared with TS only, p < .05. c Compared with TS + OCD, p < .05. d Compared with TS + ADHD, p < .05. b
course of disease. The results indicated that the cohesion, intellectual-cultural orientation, active-recreational orientation, organization and course of disease positively correlated with subjective QoL satisfaction, the conflict, achievement orientation, control negatively correlated with subjective QoL satisfaction (Table 4). To describe the linear association between subjective QoL satisfaction and a set of exploratory variables, including cohesion, intellectual-cultural orientation, active-recreational orientation, organization and course of disease conflict, achievement orientation, control, and a multiple linear regression model was developed by a stepwise method. The results indicated that the subjective QoL satisfaction was affected by exploratory variables conflict, intellectual-cultural orientation, control, cohesion, course of disease, active-recreational orientation. The exploratory
variables conflict had the biggest effect on the subjective QoL satisfaction according to the values of standardized coefficients (Table 5).
Discussion In this present study, we performed a case–control study to explore the clinical correlates of subjective QoL in a young population with TS in China. We found that in comparison with control, young people with TS engaged less in home and social activities, communicated less with adults in their family, felt more insecure about home, been over controlled and witnessed more family arguments. These findings suggested that TS could be associated with family stress and mild social withdrawal. However, this reduction in social activities was not accompanied by evidence of social anxiety.
Downloaded from jad.sagepub.com at PENNSYLVANIA STATE UNIV on November 14, 2015
5
Liu et al. Table 4. TS Subjective QoL Factor Correlation Analysis (n = 107). Measure Cohesion Conflict Achievement orientation Intellectual-cultural orientation Active-recreational orientation Organization Control Age Course of disease
M ± SD
r
p
6.65 ± 1.967 3.94 ± 2.394 6.43 ± 1.812 3.57 ± 2.270 4.10 ± 2.484 5.05 ± 2.112 3.61 ± 1.862 10.1340 ± 1.10090 2.7111 ± 1.62593
.397 −.422 −.288 .386 .222 .382 −.260 .121 .310
.000 .000 .003 .000 .022 .000 .007 .214 .001
Table 5. Multiple Linear Regression of Subjective QoL Satisfaction. Variable Constant Conflict Intellectual-cultural orientation Control Cohesion Course of disease Active-recreational orientation
β
SE
β′
t
p
45.134 −1.826 3.042 −2.865 2.907 1.832 −1.328
5.272 0.470 0.533 0.576 0.588 0.669 0.517
−0.283 0.447 −0.345 0.370 0.193 −0.214
8.561 −3.882 5.711 −4.944 4.944 2.738 −2.567
.000 .000 .000 .000 .000 .007 .012
Scores were lower for some of the QoL relationships domain (family life and cognitive component) for young people with moderate to severe tics than mild group, indicating that symptoms affect subjective QoL of children with TS. There is a certain relationship between symptoms and family life and cognitive components. Comparisons indicated that more severe tics were associated with signs of insecurity and may have a widespread negative influence on social activities within and outside the home. Another finding of this study is that even TS patients who had no co-morbidities (TS only) were showed some aspects (school life and cognitive) of QoL more negatively than controls. Preliminary research indicated that up to 88% of patients consider that having tics affects their life (Wood et al., 2003). Lower scores in the QoL school life and cognitive domain could suggest that young people with pure TS feel low self-esteem, exclusion, and so on. The result of correlation analysis indicated that the cohesion, intellectual-cultural orientation, active-recreational orientation, organization, and course of disease positively correlated with subjective QoL satisfaction, the conflict, achievement orientation, control negatively correlated with subjective QoL satisfaction. Stepwise regression analysis showed that the subjective QoL satisfaction was affected by exploratory variables conflict, intellectual-cultural orientation, control, cohesion, course of disease, active-recreational orientation (F = 19.643, p < .001). The exploratory variables conflict had the biggest effect on the subjective
QoL satisfaction according to the values of standardized coefficients. TS family environment is also an important factor that affects the subjective QoL of children with TS. Parental rearing patterns of families, physical environment, parental mood, atmosphere, and family structure have an important impact on children’s psychological development and personality formation. The contradiction between family members representing public displays of anger, aggression, and conflict degree has a negative effect on the subjective QoL of children with TS. Entertainment refers to the family members who are very loving and actively participate in social activities and a variety of recreational activities. This study showed entertainment for TS children’s subjective QoL have a positive influence on improving family environment, in order to improve the family environment. Therefore, it is necessary to reduce the contradiction and add entertainment in the family environment for TS children. Several studies have assessed the impact of TS on psychological, social, and economic aspects of well-being before the first study of QoL. Storch et al. indicated that the most frequently endorsed areas of impairment were related to school and over half of the sample reported one significant problem area due to the presence of tics, with over a third reporting two or more problem areas (Storch et al., 2007). Conelea et al. suggested that youth with chronic tic disorders (CTD) experience mild to moderate functional impairment; the impairment is generally positively correlated with tic severity, children
Downloaded from jad.sagepub.com at PENNSYLVANIA STATE UNIV on November 14, 2015
6
Journal of Attention Disorders
with CTD plus one or more co-occurring psychiatric conditions (such as ADHD, OCD) that tend to have a greater functional impairment, and a notable portion of youth with CTD experience discrimination due to tics (Conelea et al., 2011). Evaluation of QoL is a useful health monitoring tool widely used in nation and abroad. In general, subjective QoL includes emotional and cognitive components. Affective component was the emotion experience of individual in real life, such as depression, anxiety, somatic perception; cognitive component was cognitive evaluation to objective life status of individual according the accordance with its own unique standard, such as family life, school life, and social interaction. In recent years, subjective QoL of children with disease was focused by psychology and clinical workers. As a chronic recurrent disease, to fully understand the subjective QoL in children with TS and further accurate picture their impact factors have a great significance to improve the subjective QoL, disease intervention, and improve prognosis. Müller-Vahl et al. (2010) found that 57.1% of the TS group suffered from anxiety and/or depression and its subjective QoL satisfaction was significantly lower than the control group. Multivariate logistic regression analysis showed that the severity of tic symptoms and age of TS are major factor in the QoL. Compared with the control, TS children were on the lower level of subjective satisfaction in family and school life, poor self-evaluation and more negative emotions. These children suffered multiple parts of involuntary tics, involuntary abnormal sound and some bad behavior, unhappiness, inferiority, obvious depression, and lower self-evaluation. Eddy and colleagues reported that significant differences were found in TS in aspects of QoL related to home and social activities, including peer and family interactions. Patients with pure TS presented with lower QoL scores in the environment domain, poorer perceived QoL in general, and depressive features. Patients with more severe tics indicated a greater negative impact on QoL. Co-morbid OCD appeared to exert a greater impact on self and relationship QoL domains (Eddy et al., 2011). Therefore, in order to improve the QoL of children with TS, not only were the clinical symptoms actively controlled but also comprehensive psychological were intervened. To our knowledge, this was the first report on subjective QoL of TS in Chinese population. Our results indicated the subjective QoL of children with TS affected by many factors including not only clinical symptoms but also the family environment, school life, and the family conflict is the most important influencing factors. Therefore, to enhance the subjective QoL of children with TS, in addition to controlled clinical satisfaction, it should take system of psychotherapy, pay attention to improve the poor family environment, universal prevention and treatment of TS, and try to create a relaxed living environment for children, thus
further reduce the negative impact of disease on children and improve children’s QoL. Although the results of the present study are in accordance with those of previous studies, several limitations should be considered when reviewing these findings. First of all, these adults are likely to face unique challenges and impacts on QoL for which specific tools of investigation may not be sufficiently sensitive to areas that are specific to TS and a disease-specific instrument will be necessary for young people with TS. Second, the size of the sample in the current study was not relatively big. Last but not least, assessing QoL in children is a moving target due to the fact that they are constantly developing. Acknowledgment We thank all probands for their participation.
Authors’ Note Shiguo Liu and Lanlan Zheng contributed equally to this work. Mingji Yi and Xu Ma conceived and designed the experiments; Lanlan Zheng and Xueping Zheng performed the experiments; Xinhua Zhang analyzed the data; Shiguo Liu contributed analysis tools; Shiguo Liu wrote the paper.
Declaration of Conflicting Interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was supported by the National Natural Science Foundation of China (81371499 and 30971586).
References Black, K. J., & Mink, J. W. (2000). Response to levodopa challenge in Tourette syndrome. Movement Disorders, 15, 1194-1198. Bloch, M. H., Peterson, B. S., Scahill, L., Otka, J., Katsovich, L., Zhang, H., & Leckman, J. F. (2006). Adulthood outcome of tic and obsessive-compulsive symptom severity in children with Tourette syndrome. Archives of Pediatrics & Adolescent Medicine, 160, 65-69. Chang, H. L., Liang, H. Y., Wang, H. S., Li, C. S., Ko, N. C., & Hsu, Y. P. (2008). Behavioral and emotional problems in adolescents with Tourette syndrome. Chang Gung Medical Journal, 31, 145-152. Coffey, B. J., Biederman, J., Geller, D., Frazier, J., Spencer, T., Doyle, R., . . .Faraone, S. V. (2004). Reexamining Tic persistence and Tic-associated impairment in Tourette’s disorder: Findings from a naturalistic follow-up study. The Journal of Nervous and Mental Disease, 192, 776-780. Conelea, C. A., Woods, D. W., Zinner, S. H., Budman, C., Murphy, T., Scahill, L. D., . . .Walkup, J. (2011). Exploring
Downloaded from jad.sagepub.com at PENNSYLVANIA STATE UNIV on November 14, 2015
7
Liu et al. the impact of chronic tic disorders on youth: Results from the Tourette Syndrome Impact Survey. Child Psychiatry & Human Development, 42, 219-242. Dozier, J. (2006). Psychiatric and neurologic disorders that affect sleep. Respiratory Care Clinics of North America, 12, 71-80, viii. Eddy, C. M., Rizzo, R., Gulisano, M., Agodi, A., Barchitta, M., Cali, P., . . .Cavanna, A. E. (2011). Quality of life in young people with Tourette syndrome: A controlled study. Journal of Neurology, 258, 291-301. Elstner, K., Selai, C. E., Trimble, M. R., & Robertson, M. M. (2001). Quality of Life (QOL) of patients with Gilles de la Tourette’s syndrome. Acta Psychiatrica Scandinavica, 103, 52-59. Erenberg, G., Cruse, R. P., & Rothner, A. D. (1987). The natural history of Tourette syndrome: A follow-up study. Annals of Neurology, 22, 383-385. Freeman, R. D. (1997). Attention deficit hyperactivity disorder in the presence of Tourette syndrome. Neurologic Clinics, 15, 411-420. Freeman, R. D., Fast, D. K., Burd, L., Kerbeshian, J., Robertson, M. M., & Sandor, P. (2000). An international perspective on Tourette syndrome: Selected findings from 3,500 individuals in 22 countries. Developmental Medicine & Child Neurology, 42, 436-447. Kano, Y., Ohta, M., Nagai, Y., & Scahill, L. (2010). Association between Tourette syndrome and comorbidities in Japan. Brain & Development, 32, 201-207. Kurlan, R. (1992). The pathogenesis of Tourette’s syndrome. A possible role for hormonal and excitatory neurotransmitter influences in brain development. Archives of Neurology, 49, 874-876. Kurlan, R., Como, P. G., Miller, B., Palumbo, D., Deeley, C., Andresen, E. M., . . .McDermott, M. P. (2002). The behavioral spectrum of tic disorders: A community-based study. Neurology, 59, 414-420. Leckman, J. F., Bloch, M. H., King, R. A., & Scahill, L. (2006). Phenomenology of tics and natural history of tic disorders. Advances in Neurology, 99, 1-16. Leckman, J. F., Bloch, M. H., Scahill, L., & King, R. A. (2006). Tourette syndrome: The self under siege. Journal of Child Neurology, 21, 642-649. Leckman, J. F., Riddle, M. A., Hardin, M. T., Ort, S. I., Swartz, K. L., Stevenson, J., & Cohen, D. J. (1989). The Yale Global Tic Severity Scale: Initial testing of a clinician-rated scale of tic severity. Journal of the American Academy of Child & Adolescent Psychiatry, 28, 566-573. Leckman, J. F., Zhang, H., Vitale, A., Lahnin, F., Lynch, K., Bondi, C., & Peterson, B. S. (1998). Course of tic severity in Tourette syndrome: The first two decades. Pediatrics, 102, 14-19. Malone, D. A., Jr., & Pandya, M. M. (2006). Behavioral neurosurgery. Advances in Neurology, 99, 241-247. Müller-Vahl, K., Dodel, I., Muller, N., Munchau, A., Reese, J. P., Balzer-Geldsetzer, M., . . .Oertel, W. H. (2010). Healthrelated quality of life in patients with Gilles de la Tourette’s syndrome. Movement Disorders, 25, 309-314. O’Connor, K., Brisebois, H., Brault, M., Robillard, S., & Loiselle, J. (2003). Behavioral activity associated with onset in chronic
tic and habit disorder. Behaviour Research and Therapy, 41, 241-249. Pappert, E. J., Goetz, C. G., Louis, E. D., Blasucci, L., & Leurgans, S. (2003). Objective assessments of longitudinal outcome in Gilles de la Tourette’s syndrome. Neurology, 61, 936-940. Robertson, M. M. (2006). Attention deficit hyperactivity disorder, tics and Tourette’s syndrome: The relationship and treatment implications. A commentary. European Child & Adolescent Psychiatry, 15, 1-11. Singer, H. S. (2005). Tourette’s syndrome: From behaviour to biology. The Lancet Neurology, 4, 149-159. Snider, L. A., Seligman, L. D., Ketchen, B. R., Levitt, S. J., Bates, L. R., Garvey, M. A., & Swedo, S. E. (2002). Tics and problem behaviors in schoolchildren: Prevalence, characterization, and associations. Pediatrics, 110, 331-336. Storch, E. A., Lack, C. W., Simons, L. E., Goodman, W. K., Murphy, T. K., & Geffken, G. R. (2007). A measure of functional impairment in youth with Tourette’s syndrome. Journal of Pediatric Psychology, 32, 950-959. Sukhodolsky, D. G., Scahill, L., Zhang, H., Peterson, B. S., King, R. A., Lombroso, P. J., . . .Leckman, J. F. (2003). Disruptive behavior in children with Tourette’s syndrome: Association with ADHD comorbidity, tic severity, and functional impairment. Journal of the American Academy of Child & Adolescent Psychiatry, 42, 98-105. Wood, B. L., Klebba, K., Gbadebo, O., Lichter, D., Kurlan, R., & Miller, B. (2003). Pilot study of effect of emotional stimuli on tic severity in children with Tourette’s syndrome. Movement Disorders, 18, 1392-1395.
Author Biographies Shiguo Liu is a doctor in the Departments of Genetic Laboratory, The Affiliated Hospital of Medical College, Qingdao University. He received his PhD from the Peking Union Medical College, Beijing, China. Lanlan Zheng is postgraduate in the Department of psychiatry, Medical College, Qingdao University, Qingdao, China. She received her postgraduate degree in psychology from Medical College, Qingdao University, Qingdao, China. Xueping Zheng is a vice professor in the Departments of neurology, The Affiliated Hospital of Medical College, Qingdao University, Qingdao, China. She received her MD from the zhongshan University in Guangzhou, China. Xinhua Zhang is a vice professor in the Departments of Psychological clinic, The Affiliated Hospital of Medical College, Qingdao University, Qingdao, China. He received his postgraduate degree in Shanghai Jiao Tong University, China. Mingji Yi is a vice professor in the Departments of Child healthcare Department, The Affiliated Hospital of Medical College, Qingdao University, Qingdao, China. He received his postgraduate degree in psychology from Medical College, Qingdao University, Qingdao, China. Xu Ma is a professor in the Departments of genetics, National Research Institute for Family Planning, Beijing, China. His research and publications focus eugenics and medical genetics.
Downloaded from jad.sagepub.com at PENNSYLVANIA STATE UNIV on November 14, 2015
research-article2014
JADXXX10.1177/1087054713518822Journal of Attention DisordersLiu et al.
Article
The Subjective Quality of Life in Young People With Tourette Syndrome in China
Journal of Attention Disorders 1–7 © 2014 SAGE Publications Reprints and permissions: sagepub.com/journalsPermissions.nav DOI: 10.1177/1087054713518822 jad.sagepub.com
Shiguo Liu1, Lanlan Zheng1, Xueping Zheng1, Xinhua Zhang1, Mingji Yi1, and Xu Ma2,3
Abstract Objective: To explore the subjective quality of life (QoL) in children with Tourette Syndrome (TS) in China to provide a basis for more effective interference. Method: A total of 107 patients and 107 controls were enrolled. Subjective QoL was investigated by Inventory of Subjective Life Quality, Family Environment Scale of Chinese Version, and the Yale Global Tic Severity Scale, and a case–control study was performed. Results: The total score of subjective QoL and family life, school life, peer relationship, cognitive component, environment, self-awareness, cognitive component and depression experience in the TS were lower than control. Patients with co-morbid exhibited significantly lower scores within the subjective QoL family life, peer relationship, school life, self-awareness, and cognitive affective domains. Conclusion: The subjective QoL is impaired and it is important to control clinical symptoms and improve family environment for the improvement of the subjective QoL in TS. (J. of Att. Dis. XXXX; XX(X) 1-XX) Keywords Tourette syndrome, quality of life, child, China
Introduction Tourette Syndrome (TS) is a neurodevelopmental disorder characterized by motor and vocal tics. Motor tics are sudden, repetitive, stereotyped, nonrhythmic movements, while vocal or phonic tics represent involuntary sounds produced by moving air through the nose, mouth, or throat (Singer, 2005). Clinical diagnosis of TS is based on that multiple motor and one or more vocal tics are present for more than a year, and during which there was never a ticfree period of more than three consecutive months (Leckman, Bloch, King, & Scahill, 2006; Malone & Pandya, 2006). However, tics are commonly deteriorated when patients undergo emotional stimuli such as psychosocial stress, fatigue, or adversities, while they tend to ease in a state of concentrating, pleased, or asleep (O’Connor, Brisebois, Brault, Robillard, & Loiselle, 2003; Wood et al., 2003). Typically, tics usually start around 8 years of age, peak early during the second decade of life, and decline by the end of adolescence (Bloch et al., 2006; Coffey et al., 2004; Leckman et al., 1998; Pappert, Goetz, Louis, Blasucci, & Leurgans, 2003). Complete resolution occurs by age 18 in 50% of patients (Leckman, Bloch, Scahill, & King, 2006), and only 20% or fewer of children with TS continue to present a moderate level of impairment of global functioning by the age of 20. Clinic data showed that males are five times more likely to be affected than females, and the prevalence may approach 0.72% (Dozier, 2006).
Although the hallmark of TS is the presence of motor and vocal tics, the clinical spectrum also includes a variety of psychiatric co-morbidities. Approximately 90% of TS patients experience a psychiatric co-morbidity (Kurlan et al., 2002), and the most common form is the ADHD (Freeman, 1997; Robertson, 2006). Around 65% of children with TS are highly associated with ADHD and the clinical spectrum of the two neurodevelopmental disorders tends to overlap (Sukhodolsky et al., 2003). This co-occurrence of TS and ADHD can be associated with disruptive behaviors such as aggression, explosive behavior, low frustration tolerance, and noncompliance (Black & Mink, 2000; Kurlan et al., 2002; Snider et al., 2002). The obsessive-compulsive disorder (OCD) is another common co-morbidity for TS. Over half of TS patients also have prominent OCD. Kano and co-workers reported the association between TS and co-morbidities in Japan, in which TS patients’ co-concurrence with OCD showed significant higher frequency for coprophenomana, impulsiveness, school refusal, self-injurious behaviors, and 1
Qingdao University, China Peking Union Medical College, Beijing, China 3 National Research Institute for Family Planning, Beijing, China 2
Corresponding Author: Mingji Yi, Child Healthcare Department, The Affiliated Hospital of Medical College, Qingdao University, 16 Jiangsu Road, Qingdao 266003, China. Email: [email protected]
Downloaded from jad.sagepub.com at PENNSYLVANIA STATE UNIV on November 14, 2015
2
Journal of Attention Disorders
clumsiness than that of patients with TS only (Kano, Ohta, Nagai, & Scahill, 2010). They further demonstrated that tic symptoms and impairment during the worst period were significantly severer in TS patients with OCD than that of patients with TS only. Other co-morbidities may also be apparent such as phobias, anxiety, aggressive, oppositional defiant disorder, depression, and other mood disorders (Kano et al., 2010). As preliminary research showed that up to 88% of patients consider that having tics interferes their life, considerable attention has been devoted to the study of subjective quality of life (QoL) in those people (Erenberg, Cruse, & Rothner, 1987). Perceived subjective QoL can determine the subjective experience of living with a health condition, affect planning for the future, and potentially affect acceptance of and adherence to treatment. There is a need to consider the features of TS that have the most significant impact on young people’s subjective QoL, so that clinicians can manage these effectively. The first study of QoL of patients with TS demonstrated that patients with TS showed significantly worse QoL than a general population sample (Elstner, Selai, Trimble, & Obertson, 2001). It is also reported that the subjective QoL of young people with tics in the United States was lower than that of healthy controls with respect to emotional, social, and school functioning but comparable in terms of physical functioning. Studies also showed that tic severity was inversely related to social acceptableness. The purpose of the present study was to evaluate subjective QoL in a large sample of young patients with TS in China from specialized outpatient clinics. We also analyzed whether tic severity or co-morbid (OCD and/or ADHD) impair the subjective QoL of patients with TS.
Material and Method Participants Participants were recruited from Department of Child Health Care, Affiliated Hospital of Qingdao University Medical College from 2008 until 2011. Participants included 107 young people with TS (92 males, 15 females), average age is M = 10.1 ± 1.1 and 107 control (59 males, 48 females), average age is 9.9 ± 1.0. The diagnoses of TS and other clinical conditions were made in accordance with Diagnostic and Statistical Manual of Mental Disorders (4th ed., text rev.; DSM-IV-TR; American Psychiatric Association, 2000) criteria by a psychiatrist with extensive experience. The control group had no psychiatric disorders, which undergo the same diagnostic evaluations that the TS group participants did.
Measures Inventory of Subjective Life Quality (ISLQ). The ISLQ (Freeman et al., 2000) evaluate two components (cognitive as
Points and the affective component) and eight dimensions (family life, peer relationship, School life, living environment, self-knowledge, the experience of depression, the experience of anxiety and physical emotion). The composition, dimensions and total score higher, indicating higher levels of satisfaction. Family Environment Scale of Chinese Version (FES-CV). FES was prepared in 1981, 90 questions, all of them are true or false, and takes about 30 min to complete (Kurlan, 1992). The scale is divided into 10 subscales; respectively evaluate 10 different families, social and environmental characteristics. In many countries, the FES has been widely used to describe different types of family characteristics and the family situation in state of crisis, evaluate the home environment changes in family intervention, as well as the comparison of other aspects of family environment and family life. FES’s evaluation of household characteristics include cohesion, expressiveness, conflict, independence, achievement orientation, intellectual-cultural orientation, active-recreational orientation, moral-religious emphasis, organization, and control. FES need to be modified to use in China, this is because some of the concepts assessed in the scale is not suitable for Chinese families, the contents of several projects in the Chinese cultural environment seem very inappropriate (such as independence, moral-religious emphasis, and expressiveness). Therefore, we used FES-CV and excluded three items (independence, moral-religious emphasis, and expressiveness). The Yale Global Tic Severity Scale (YGTSS). The YGTSS is a reliable clinician-rated interview (Leckman et al., 1989). The clinician initially notes the presence of motor and phonic tics based on child and parent reports over the past week, as well as behavioral observations. Then, the clinician rates the severity of motor and phonic tics on five separate dimensions each: number, frequency, intensity, complexity, and interference. Five index scores are obtained: Total Motor Tic Score, Total Phonic Tic Score, Total Tic Score, Overall Impairment Rating, and Global Severity Score. According to Global Severity Score, the severities of tic symptoms are divided into three grades: mild (50 points; Chang et al., 2008).
Statistics Data were analyzed using SPSS software, PC version 17.0 for Windows. Quantitative data were expressed by the means ± standard deviations (M ± SD), and group means were compared by t test and ANOVA. The first part of analysis described the general information of TS children and control children; the second part of analysis compared the subjective QoL satisfaction scores given by patients with TS to control. The third part of analysis investigated whether patients with TS who had more severe tics or co-morbid
Downloaded from jad.sagepub.com at PENNSYLVANIA STATE UNIV on November 14, 2015
3
Liu et al. Table 1. General Condition of TS Children and Control Children. Task
Category
Control
TS
χ2
p
Sex
man Female 2,500-4,000 kg Other Enlarge core Core Other Medium Other Junior and below High school College and above Junior and below High school College and above First child Second child and more Breast milk only Breast milk mainly Other
59 48 96 27 34 63 10 99 8 63 20
92 15 110 16 18 73 16 65 42 39 35 33 57 20 30 77 30 65 12 30
24.498
.000
3.730
.065
7.043
.030
30.169
.000
11.159
.004
2.620
.270
0.357
.654
25.602
.000
Born weight Family Household income Father’s education Mother’s education Children parity Feeding situation (4 months)
exhibited greater differences than the control groups on the measures administered. The last part of analysis explores the influencing factors of the subjective QoL satisfaction in children with TS.
Results Study of General Information of TS Children and Control Children There are significant difference between two groups of children in the sex ratio, family structure, father’s educational background, birth weight, family income, and feeding situation (4 months; p < .05 or p < .01), and no significant difference between two groups of children in birth order and mother’s educational background (Table 1).
Subjective QoL for Control and TS The second part of the analysis compared the subjective QoL scores given by patients with TS to control. Patients with TS scored significantly lower than control for family life, Peer relationship, School life, Environment, Selfawareness, Experience depression, Cognitive component, and total (p < .05; Table 2).
Subjective QoL for Severity Scale of TS First, we split patients with TS into two groups: mild and moderate to severe. Patients with moderate to severe had
24 22 20 73 34 57 40 10
significantly lower scores than milder TS for family life and cognitive component (p < 0.05). The second was dependent on the presence of co-morbidities, which generated five subgroups. The subgroups were independently compared with each other for scores on the scales administered. Subjective QoL may be more affected children with TS who suffer with other co-morbid conditions such as OCD and ADHD. The group of TS was split into four subgroups based on the presence of co-morbid conditions, so that they could be separately compared with controls. The subgroups were as follows: TS only (n = 52); TS plus OCD (TS + OCD: n = 9); TS plus ADHD (TS + ADHD: n = 34); TS plus OCD and ADHD (TS + OCD + ADHD: n = 12; Table 3). Patients with TS + OCD + ADHD exhibited significantly lower total subjective QoL score than control and significantly lower scores within the subjective QoL family life, peer relationship, school life, self-awareness, cognitive affective domains. These findings indicate that young people with TS + OCD + ADHD may experience social difficulties because of their symptoms and are perhaps less likely to spend time socializing with adults in the family at home.
Subjective QoL Factor Correlation Analysis Data from patients with TS were collected to investigate the correlation between factors and subjective QoL satisfaction score. The factors contain three feathers of Family Environment Scale, the age of patients, behavior score, and
Downloaded from jad.sagepub.com at PENNSYLVANIA STATE UNIV on November 14, 2015
4
Journal of Attention Disorders
Table 2. Mean and SD for Statistically Different Scores on the Subjective QoL for Control and TS. Measure Family life Peer relationship School life Environment Self-awareness Experience depression Anxiety experience The body emotion Cognitive component Affective component Total
Control (n = 107)
TS (n = 107)
t
p
7.19 ± 0.892 6.72 ± 1.559 6.31 ± 0.915 6.71 ± 1.402 6.39 ± 1.155 6.19 ± 1.894 5.63 ± 1.794 5.48 ± 2.2.107 68.69 ± 11.997 57.05 ± 18.281 65.97 ± 14.070
6.72 ± 1.026 6.07 ± 1.592 5.38 ± 1.146 6.14 ± 2.238 5.81 ± 1.573 5.47 ± 2.466 5.91 ± 2.196 4.87 ± 2.128 59.52 ± 11.942 56.05 ± 21.185 57.32 ± 15.447
3.556 3.037 6.524 2.891 3.071 2.394 −1.023 2.098 5.603 0.367 4.284
.000 .003 .000 .004 .002 .018 .308 .607 .000 .714 .000
Table 3. Mean and SD for Statistically Different Scores on the Subjective QoL for Control and the Co-Morbidity Subgroups. Measure Family life Peer relationship School life Environment Self-awareness Depression Anxiety Body emotion Cognitive Affective Total
Control (n = 107) 7.19 ± 0.892 6.72 ± 1.559 6.31 ± 0.915 6.21 ± 2.227 6.39 ± 1.155 6.19 ± 1.894 5.63 ± 1.794 5.48 ± 2.107 68.69 ± 11.997 57.05 ± 18.281 65.97 ± 14.070
TS only (n = 52) 7.17 ± 1.150 6.50 ± 1.639 5.38 ± 1.191a 6.38 ± 1.430 6.37 ± 1.442 5.85 ± 2.461 6.23 ± 2.454 4.94 ± 2.492 63.92 ± 12.811a 58.44 ± 26.312 62.10 ± 17.882
TS + OCD (n = 9)
TS + ADHD (n = 34) a,b
6.33 ± 1.000 5.89 ± 1.764 5.89 ± 0.928 5.78 ± 1.302 7.22 ± 1.202 6.56 ± 1.333 7.56 ± 1.333 4.67 ± 1.323 62.78 ± 9.770 67.89 ± 10.043 65.22 ± 6.360
TS + OCD + ADHD (n = 12)
a,b
6.47 ± 0.507 5.82 ± 1.507a,b 5.38 ± 1.231a 6.09 ± 1.240 4.97 ± 0.937a,b,c 5.38 ± 2.104 5.29 ± 1.697b,c 4.59 ± 1.811a 55.91 ± 7.921a,b 50.29 ± 13.543c 52.71 ± 9.114a,b,c
a,b,d
5.75 ± 0.452 5.00 ± 0.739a,b 5.00 ± 0.739a 5.50 ± 1.732 4.75 ± 2.006a,b,c 3.25 ± 2.989a,b,c,d 5.00 ± 1.954c 5.50 ± 1.732 48.25 ± 8.750a,b,c,d 43.75 ± 23.913a,b,c 43.75 ± 10.610a,b,c
F
p
11.377 5.075 11.628 0.685 14.054 5.794 3.709 1.543 14.482 2.870 10.915
.000 .001 .000 .603 .000 .000 .006 .191 .000 .024 .000
a
Compared with control, p < .05. Compared with TS only, p < .05. c Compared with TS + OCD, p < .05. d Compared with TS + ADHD, p < .05. b
course of disease. The results indicated that the cohesion, intellectual-cultural orientation, active-recreational orientation, organization and course of disease positively correlated with subjective QoL satisfaction, the conflict, achievement orientation, control negatively correlated with subjective QoL satisfaction (Table 4). To describe the linear association between subjective QoL satisfaction and a set of exploratory variables, including cohesion, intellectual-cultural orientation, active-recreational orientation, organization and course of disease conflict, achievement orientation, control, and a multiple linear regression model was developed by a stepwise method. The results indicated that the subjective QoL satisfaction was affected by exploratory variables conflict, intellectual-cultural orientation, control, cohesion, course of disease, active-recreational orientation. The exploratory
variables conflict had the biggest effect on the subjective QoL satisfaction according to the values of standardized coefficients (Table 5).
Discussion In this present study, we performed a case–control study to explore the clinical correlates of subjective QoL in a young population with TS in China. We found that in comparison with control, young people with TS engaged less in home and social activities, communicated less with adults in their family, felt more insecure about home, been over controlled and witnessed more family arguments. These findings suggested that TS could be associated with family stress and mild social withdrawal. However, this reduction in social activities was not accompanied by evidence of social anxiety.
Downloaded from jad.sagepub.com at PENNSYLVANIA STATE UNIV on November 14, 2015
5
Liu et al. Table 4. TS Subjective QoL Factor Correlation Analysis (n = 107). Measure Cohesion Conflict Achievement orientation Intellectual-cultural orientation Active-recreational orientation Organization Control Age Course of disease
M ± SD
r
p
6.65 ± 1.967 3.94 ± 2.394 6.43 ± 1.812 3.57 ± 2.270 4.10 ± 2.484 5.05 ± 2.112 3.61 ± 1.862 10.1340 ± 1.10090 2.7111 ± 1.62593
.397 −.422 −.288 .386 .222 .382 −.260 .121 .310
.000 .000 .003 .000 .022 .000 .007 .214 .001
Table 5. Multiple Linear Regression of Subjective QoL Satisfaction. Variable Constant Conflict Intellectual-cultural orientation Control Cohesion Course of disease Active-recreational orientation
β
SE
β′
t
p
45.134 −1.826 3.042 −2.865 2.907 1.832 −1.328
5.272 0.470 0.533 0.576 0.588 0.669 0.517
−0.283 0.447 −0.345 0.370 0.193 −0.214
8.561 −3.882 5.711 −4.944 4.944 2.738 −2.567
.000 .000 .000 .000 .000 .007 .012
Scores were lower for some of the QoL relationships domain (family life and cognitive component) for young people with moderate to severe tics than mild group, indicating that symptoms affect subjective QoL of children with TS. There is a certain relationship between symptoms and family life and cognitive components. Comparisons indicated that more severe tics were associated with signs of insecurity and may have a widespread negative influence on social activities within and outside the home. Another finding of this study is that even TS patients who had no co-morbidities (TS only) were showed some aspects (school life and cognitive) of QoL more negatively than controls. Preliminary research indicated that up to 88% of patients consider that having tics affects their life (Wood et al., 2003). Lower scores in the QoL school life and cognitive domain could suggest that young people with pure TS feel low self-esteem, exclusion, and so on. The result of correlation analysis indicated that the cohesion, intellectual-cultural orientation, active-recreational orientation, organization, and course of disease positively correlated with subjective QoL satisfaction, the conflict, achievement orientation, control negatively correlated with subjective QoL satisfaction. Stepwise regression analysis showed that the subjective QoL satisfaction was affected by exploratory variables conflict, intellectual-cultural orientation, control, cohesion, course of disease, active-recreational orientation (F = 19.643, p < .001). The exploratory variables conflict had the biggest effect on the subjective
QoL satisfaction according to the values of standardized coefficients. TS family environment is also an important factor that affects the subjective QoL of children with TS. Parental rearing patterns of families, physical environment, parental mood, atmosphere, and family structure have an important impact on children’s psychological development and personality formation. The contradiction between family members representing public displays of anger, aggression, and conflict degree has a negative effect on the subjective QoL of children with TS. Entertainment refers to the family members who are very loving and actively participate in social activities and a variety of recreational activities. This study showed entertainment for TS children’s subjective QoL have a positive influence on improving family environment, in order to improve the family environment. Therefore, it is necessary to reduce the contradiction and add entertainment in the family environment for TS children. Several studies have assessed the impact of TS on psychological, social, and economic aspects of well-being before the first study of QoL. Storch et al. indicated that the most frequently endorsed areas of impairment were related to school and over half of the sample reported one significant problem area due to the presence of tics, with over a third reporting two or more problem areas (Storch et al., 2007). Conelea et al. suggested that youth with chronic tic disorders (CTD) experience mild to moderate functional impairment; the impairment is generally positively correlated with tic severity, children
Downloaded from jad.sagepub.com at PENNSYLVANIA STATE UNIV on November 14, 2015
6
Journal of Attention Disorders
with CTD plus one or more co-occurring psychiatric conditions (such as ADHD, OCD) that tend to have a greater functional impairment, and a notable portion of youth with CTD experience discrimination due to tics (Conelea et al., 2011). Evaluation of QoL is a useful health monitoring tool widely used in nation and abroad. In general, subjective QoL includes emotional and cognitive components. Affective component was the emotion experience of individual in real life, such as depression, anxiety, somatic perception; cognitive component was cognitive evaluation to objective life status of individual according the accordance with its own unique standard, such as family life, school life, and social interaction. In recent years, subjective QoL of children with disease was focused by psychology and clinical workers. As a chronic recurrent disease, to fully understand the subjective QoL in children with TS and further accurate picture their impact factors have a great significance to improve the subjective QoL, disease intervention, and improve prognosis. Müller-Vahl et al. (2010) found that 57.1% of the TS group suffered from anxiety and/or depression and its subjective QoL satisfaction was significantly lower than the control group. Multivariate logistic regression analysis showed that the severity of tic symptoms and age of TS are major factor in the QoL. Compared with the control, TS children were on the lower level of subjective satisfaction in family and school life, poor self-evaluation and more negative emotions. These children suffered multiple parts of involuntary tics, involuntary abnormal sound and some bad behavior, unhappiness, inferiority, obvious depression, and lower self-evaluation. Eddy and colleagues reported that significant differences were found in TS in aspects of QoL related to home and social activities, including peer and family interactions. Patients with pure TS presented with lower QoL scores in the environment domain, poorer perceived QoL in general, and depressive features. Patients with more severe tics indicated a greater negative impact on QoL. Co-morbid OCD appeared to exert a greater impact on self and relationship QoL domains (Eddy et al., 2011). Therefore, in order to improve the QoL of children with TS, not only were the clinical symptoms actively controlled but also comprehensive psychological were intervened. To our knowledge, this was the first report on subjective QoL of TS in Chinese population. Our results indicated the subjective QoL of children with TS affected by many factors including not only clinical symptoms but also the family environment, school life, and the family conflict is the most important influencing factors. Therefore, to enhance the subjective QoL of children with TS, in addition to controlled clinical satisfaction, it should take system of psychotherapy, pay attention to improve the poor family environment, universal prevention and treatment of TS, and try to create a relaxed living environment for children, thus
further reduce the negative impact of disease on children and improve children’s QoL. Although the results of the present study are in accordance with those of previous studies, several limitations should be considered when reviewing these findings. First of all, these adults are likely to face unique challenges and impacts on QoL for which specific tools of investigation may not be sufficiently sensitive to areas that are specific to TS and a disease-specific instrument will be necessary for young people with TS. Second, the size of the sample in the current study was not relatively big. Last but not least, assessing QoL in children is a moving target due to the fact that they are constantly developing. Acknowledgment We thank all probands for their participation.
Authors’ Note Shiguo Liu and Lanlan Zheng contributed equally to this work. Mingji Yi and Xu Ma conceived and designed the experiments; Lanlan Zheng and Xueping Zheng performed the experiments; Xinhua Zhang analyzed the data; Shiguo Liu contributed analysis tools; Shiguo Liu wrote the paper.
Declaration of Conflicting Interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was supported by the National Natural Science Foundation of China (81371499 and 30971586).
References Black, K. J., & Mink, J. W. (2000). Response to levodopa challenge in Tourette syndrome. Movement Disorders, 15, 1194-1198. Bloch, M. H., Peterson, B. S., Scahill, L., Otka, J., Katsovich, L., Zhang, H., & Leckman, J. F. (2006). Adulthood outcome of tic and obsessive-compulsive symptom severity in children with Tourette syndrome. Archives of Pediatrics & Adolescent Medicine, 160, 65-69. Chang, H. L., Liang, H. Y., Wang, H. S., Li, C. S., Ko, N. C., & Hsu, Y. P. (2008). Behavioral and emotional problems in adolescents with Tourette syndrome. Chang Gung Medical Journal, 31, 145-152. Coffey, B. J., Biederman, J., Geller, D., Frazier, J., Spencer, T., Doyle, R., . . .Faraone, S. V. (2004). Reexamining Tic persistence and Tic-associated impairment in Tourette’s disorder: Findings from a naturalistic follow-up study. The Journal of Nervous and Mental Disease, 192, 776-780. Conelea, C. A., Woods, D. W., Zinner, S. H., Budman, C., Murphy, T., Scahill, L. D., . . .Walkup, J. (2011). Exploring
Downloaded from jad.sagepub.com at PENNSYLVANIA STATE UNIV on November 14, 2015
7
Liu et al. the impact of chronic tic disorders on youth: Results from the Tourette Syndrome Impact Survey. Child Psychiatry & Human Development, 42, 219-242. Dozier, J. (2006). Psychiatric and neurologic disorders that affect sleep. Respiratory Care Clinics of North America, 12, 71-80, viii. Eddy, C. M., Rizzo, R., Gulisano, M., Agodi, A., Barchitta, M., Cali, P., . . .Cavanna, A. E. (2011). Quality of life in young people with Tourette syndrome: A controlled study. Journal of Neurology, 258, 291-301. Elstner, K., Selai, C. E., Trimble, M. R., & Robertson, M. M. (2001). Quality of Life (QOL) of patients with Gilles de la Tourette’s syndrome. Acta Psychiatrica Scandinavica, 103, 52-59. Erenberg, G., Cruse, R. P., & Rothner, A. D. (1987). The natural history of Tourette syndrome: A follow-up study. Annals of Neurology, 22, 383-385. Freeman, R. D. (1997). Attention deficit hyperactivity disorder in the presence of Tourette syndrome. Neurologic Clinics, 15, 411-420. Freeman, R. D., Fast, D. K., Burd, L., Kerbeshian, J., Robertson, M. M., & Sandor, P. (2000). An international perspective on Tourette syndrome: Selected findings from 3,500 individuals in 22 countries. Developmental Medicine & Child Neurology, 42, 436-447. Kano, Y., Ohta, M., Nagai, Y., & Scahill, L. (2010). Association between Tourette syndrome and comorbidities in Japan. Brain & Development, 32, 201-207. Kurlan, R. (1992). The pathogenesis of Tourette’s syndrome. A possible role for hormonal and excitatory neurotransmitter influences in brain development. Archives of Neurology, 49, 874-876. Kurlan, R., Como, P. G., Miller, B., Palumbo, D., Deeley, C., Andresen, E. M., . . .McDermott, M. P. (2002). The behavioral spectrum of tic disorders: A community-based study. Neurology, 59, 414-420. Leckman, J. F., Bloch, M. H., King, R. A., & Scahill, L. (2006). Phenomenology of tics and natural history of tic disorders. Advances in Neurology, 99, 1-16. Leckman, J. F., Bloch, M. H., Scahill, L., & King, R. A. (2006). Tourette syndrome: The self under siege. Journal of Child Neurology, 21, 642-649. Leckman, J. F., Riddle, M. A., Hardin, M. T., Ort, S. I., Swartz, K. L., Stevenson, J., & Cohen, D. J. (1989). The Yale Global Tic Severity Scale: Initial testing of a clinician-rated scale of tic severity. Journal of the American Academy of Child & Adolescent Psychiatry, 28, 566-573. Leckman, J. F., Zhang, H., Vitale, A., Lahnin, F., Lynch, K., Bondi, C., & Peterson, B. S. (1998). Course of tic severity in Tourette syndrome: The first two decades. Pediatrics, 102, 14-19. Malone, D. A., Jr., & Pandya, M. M. (2006). Behavioral neurosurgery. Advances in Neurology, 99, 241-247. Müller-Vahl, K., Dodel, I., Muller, N., Munchau, A., Reese, J. P., Balzer-Geldsetzer, M., . . .Oertel, W. H. (2010). Healthrelated quality of life in patients with Gilles de la Tourette’s syndrome. Movement Disorders, 25, 309-314. O’Connor, K., Brisebois, H., Brault, M., Robillard, S., & Loiselle, J. (2003). Behavioral activity associated with onset in chronic
tic and habit disorder. Behaviour Research and Therapy, 41, 241-249. Pappert, E. J., Goetz, C. G., Louis, E. D., Blasucci, L., & Leurgans, S. (2003). Objective assessments of longitudinal outcome in Gilles de la Tourette’s syndrome. Neurology, 61, 936-940. Robertson, M. M. (2006). Attention deficit hyperactivity disorder, tics and Tourette’s syndrome: The relationship and treatment implications. A commentary. European Child & Adolescent Psychiatry, 15, 1-11. Singer, H. S. (2005). Tourette’s syndrome: From behaviour to biology. The Lancet Neurology, 4, 149-159. Snider, L. A., Seligman, L. D., Ketchen, B. R., Levitt, S. J., Bates, L. R., Garvey, M. A., & Swedo, S. E. (2002). Tics and problem behaviors in schoolchildren: Prevalence, characterization, and associations. Pediatrics, 110, 331-336. Storch, E. A., Lack, C. W., Simons, L. E., Goodman, W. K., Murphy, T. K., & Geffken, G. R. (2007). A measure of functional impairment in youth with Tourette’s syndrome. Journal of Pediatric Psychology, 32, 950-959. Sukhodolsky, D. G., Scahill, L., Zhang, H., Peterson, B. S., King, R. A., Lombroso, P. J., . . .Leckman, J. F. (2003). Disruptive behavior in children with Tourette’s syndrome: Association with ADHD comorbidity, tic severity, and functional impairment. Journal of the American Academy of Child & Adolescent Psychiatry, 42, 98-105. Wood, B. L., Klebba, K., Gbadebo, O., Lichter, D., Kurlan, R., & Miller, B. (2003). Pilot study of effect of emotional stimuli on tic severity in children with Tourette’s syndrome. Movement Disorders, 18, 1392-1395.
Author Biographies Shiguo Liu is a doctor in the Departments of Genetic Laboratory, The Affiliated Hospital of Medical College, Qingdao University. He received his PhD from the Peking Union Medical College, Beijing, China. Lanlan Zheng is postgraduate in the Department of psychiatry, Medical College, Qingdao University, Qingdao, China. She received her postgraduate degree in psychology from Medical College, Qingdao University, Qingdao, China. Xueping Zheng is a vice professor in the Departments of neurology, The Affiliated Hospital of Medical College, Qingdao University, Qingdao, China. She received her MD from the zhongshan University in Guangzhou, China. Xinhua Zhang is a vice professor in the Departments of Psychological clinic, The Affiliated Hospital of Medical College, Qingdao University, Qingdao, China. He received his postgraduate degree in Shanghai Jiao Tong University, China. Mingji Yi is a vice professor in the Departments of Child healthcare Department, The Affiliated Hospital of Medical College, Qingdao University, Qingdao, China. He received his postgraduate degree in psychology from Medical College, Qingdao University, Qingdao, China. Xu Ma is a professor in the Departments of genetics, National Research Institute for Family Planning, Beijing, China. His research and publications focus eugenics and medical genetics.
Downloaded from jad.sagepub.com at PENNSYLVANIA STATE UNIV on November 14, 2015
