Neuropsychiatric symptoms in siblings of children with Tourette syndrome in the EMTICS study

IF 3.1
JCPP advances Pub Date : 2024-09-20 DOI:10.1002/jcv2.12277
Olga Sidiropoulou, Jennifer Glaus, Julie Hagstrøm, Setareh Ranjbar, Renata Rizzo, EMTICS collaborative group, Pieter J. Hoekstra, Andrea Dietrich, Kerstin J. Plessen
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Abstract

Background

Tourette syndrome (TS) is associated with neuropsychiatric comorbidities, such as autism spectrum disorder (ASD), attention-deficit/hyperactivity disorder (ADHD) and oppositional defiant disorder (ODD). Even though comorbidities are the main source of impairment in individuals with TS, family aggregation between TS and other neuropsychiatric disorders has been little explored. We therefore investigated associations of tic severity in probands with symptoms of ASD, ADHD, and ODD in their siblings and the influence of tic severity, age, and sex.

Methods

The sample of the present study stems from the European Multicenter Tics in Children Study (EMTICS), a longitudinal observational study, with the present subsample of 196 probands with TS and their 220 full siblings (54.1% girls). We analyzed associations of probands’ tic severity with ASD, ADHD, and ODD symptoms in their siblings using generalized linear mixed-effect negative binomial regression models.

Results

Higher tic severity in probands was associated with higher scores of ASD symptoms in their siblings (IRR = 1.48, 95% confidence interval [95% CI] 1.03–2.12, p = 0.034); after excluding the three items in the Autism Spectrum Screening Questionnaire linked to stereotypies (that may be misinterpreted as tic-like behaviors; IRR = 1.44 [95% CI] 0.99–2.09, p = 0.057) the effect size remained similar, yet reaching only near-significance. Moreover, we demonstrated a significant interaction between probands’ tic severity and sex upon siblings’ symptoms of ADHD and ODD. Female siblings of probands with higher tic severity displayed more symptoms of ADHD and ODD, whereas this effect was absent in male siblings.

Conclusions

This multicenter study demonstrated a link between probands’ current tic severity and siblings’ neuropsychiatric symptoms. Our study suggests a familial link between TS and ASD-like symptoms, competencies as well as sex-specific associations with ADHD and ODD symptoms in female siblings. The current study sheds light on a broader family tendency and highlights the need for targeted prevention in this vulnerable population. Our findings, however, call for further studies to better understand the genetic and environmental aggregation of influences between individuals with TS, ADHD, and ODD and their siblings.

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EMTICS研究中图雷特综合征患儿兄弟姐妹的神经精神症状
图雷特综合征(TS)与神经精神合并症有关,如自闭症谱系障碍(ASD)、注意缺陷/多动障碍(ADHD)和对立违抗性障碍(ODD)。尽管合并症是TS患者损伤的主要来源,但TS与其他神经精神疾病之间的家族聚集性研究很少。因此,我们调查了先证者的抽动严重程度与其兄弟姐妹的ASD、ADHD和ODD症状的关系,以及抽动严重程度、年龄和性别的影响。方法本研究的样本来源于欧洲多中心儿童抽搐研究(EMTICS),这是一项纵向观察研究,目前的子样本为196名TS先证者及其220名全兄妹(54.1%为女孩)。我们使用广义线性混合效应负二项回归模型分析先证的抽动严重程度与其兄弟姐妹的ASD、ADHD和ODD症状的关联。结果先证者抽动严重程度越高,其兄弟姐妹的ASD症状评分越高(IRR = 1.48, 95%可信区间[95% CI] 1.03-2.12, p = 0.034);在排除自闭症谱系筛查问卷中与刻板印象(可能被误解为类抽动行为;IRR = 1.44 [95% CI] 0.99-2.09, p = 0.057)相关的三个项目后,效应量仍然相似,但仅接近显著性。此外,我们证明先证者的抽搐严重程度和性别对兄弟姐妹的ADHD和ODD症状有显著的相互作用。抽动严重程度较高的先证女性兄弟姐妹表现出更多的ADHD和ODD症状,而这种影响在男性兄弟姐妹中不存在。结论:这项多中心研究表明先证者当前抽动严重程度与兄弟姐妹的神经精神症状之间存在联系。我们的研究表明,在女性兄弟姐妹中,TS和asd样症状、能力以及与ADHD和ODD症状的性别特异性关联之间存在家族联系。目前的研究揭示了更广泛的家庭倾向,并强调了在这一弱势群体中进行有针对性预防的必要性。然而,我们的发现需要进一步的研究,以更好地了解TS、ADHD和ODD个体及其兄弟姐妹之间的遗传和环境影响。
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