The microstructural change of the brain and its clinical severity association in pediatric Tourette syndrome patients.

IF 4.1 2区 医学 Q1 CLINICAL NEUROLOGY Journal of Neurodevelopmental Disorders Pub Date : 2023-10-25 DOI:10.1186/s11689-023-09501-0
Chia-Jui Hsu, Lee Chin Wong, Hsin-Pei Wang, Yi-Chun Chung, Te-Wei Kao, Chen-Hsiang Weng, Wen-Chau Wu, Shinn-Forng Peng, Wen-Yih Isaac Tseng, Wang-Tso Lee
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Abstract

Background: Gilles de la Tourette syndrome (GTS) is a prevalent pediatric neurological disorder. Most studies point to abnormalities in the cortico-striato-thalamocortical (CSTC) circuits. Neuroimaging studies have shown GTS's extensive impact on the entire brain. However, due to participant variability and potential drug and comorbidity impact, the results are inconsistent. To mitigate the potential impact of participant heterogeneity, we excluded individuals with comorbidities or those currently undergoing medication treatments. Based on the hypothesis of abnormality within the CSTC circuit, we investigated microstructural changes in white matter using diffusion spectrum imaging (DSI). This study offers the first examination of microstructural changes in treatment-naïve pediatric patients with pure GTS using diffusion spectrum imaging.

Methods: This single-center prospective study involved 30 patients and 30 age- and gender-matched healthy volunteers who underwent sagittal T1-weighted MRI and DSI. We analyzed generalized fractional anisotropy, mean diffusivity, axial diffusivity, and radial diffusivity.

Results: No significant differences were observed in mean diffusivity and axial diffusivity values between the two groups. However, the patient group exhibited significantly higher generalized fractional anisotropy values in the right frontostriatal tract of the dorsolateral prefrontal cortex, the right frontostriatal tract of the precentral gyrus, and bilateral thalamic radiation of the dorsolateral prefrontal cortex. Additionally, the generalized fractional anisotropy value of the right frontostriatal tract of the precentral gyrus is inversely correlated with the total tic severity scores at the most severe condition.

Conclusion: Treatment-naïve pediatric GTS patients demonstrated increased connectivity within the CSTC circuit as per diffusion spectrum imaging, indicating possible CSTC circuit dysregulation. This finding could also suggest a compensatory change. It thus underscores the necessity of further investigation into the fundamental pathological changes in GTS. Nevertheless, the observed altered connectivity in GTS patients might serve as a potential target for therapeutic intervention.

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儿童抽动秽语综合征患者大脑的微观结构变化及其临床严重程度的相关性。
背景:抽动秽语综合征(GTS)是一种常见的儿科神经系统疾病。大多数研究都指出皮质-纹状体-丘脑皮质(CSTC)回路异常。神经影像学研究表明GTS对整个大脑有广泛的影响。然而,由于参与者的变异性以及潜在的药物和共病影响,结果并不一致。为了减轻参与者异质性的潜在影响,我们排除了患有合并症的个体或目前正在接受药物治疗的个体。基于CSTC电路异常的假设,我们使用扩散光谱成像(DSI)研究了白质的微观结构变化。这项研究首次使用扩散光谱成像检查了患有纯GTS的治疗幼稚儿童患者的微观结构变化。方法:这项单中心前瞻性研究涉及30名患者和30名年龄和性别匹配的健康志愿者,他们接受了矢状T1加权MRI和DSI检查。我们分析了广义分数各向异性、平均扩散率、轴向扩散率和径向扩散率。结果:两组之间的平均扩散率和轴向扩散率值没有显著差异。然而,患者组在背外侧前额叶皮层的右额纹状体束、中央前回的右额丘脑束和背外侧前皮层的双侧丘脑辐射中表现出显著更高的广义各向异性分数值。此外,中央前回右额纹状体束的广义各向异性分数值与最严重情况下的抽动严重程度总分呈负相关。结论:根据扩散光谱成像,治疗幼稚的儿童GTS患者表现出CSTC回路内的连接增加,表明CSTC回路可能失调。这一发现也可能预示着一种补偿性的变化。因此,它强调了进一步研究GTS的基本病理变化的必要性。然而,在GTS患者中观察到的连接改变可能是治疗干预的潜在目标。
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来源期刊
CiteScore
7.60
自引率
4.10%
发文量
58
审稿时长
>12 weeks
期刊介绍: Journal of Neurodevelopmental Disorders is an open access journal that integrates current, cutting-edge research across a number of disciplines, including neurobiology, genetics, cognitive neuroscience, psychiatry and psychology. The journal’s primary focus is on the pathogenesis of neurodevelopmental disorders including autism, fragile X syndrome, tuberous sclerosis, Turner Syndrome, 22q Deletion Syndrome, Prader-Willi and Angelman Syndrome, Williams syndrome, lysosomal storage diseases, dyslexia, specific language impairment and fetal alcohol syndrome. With the discovery of specific genes underlying neurodevelopmental syndromes, the emergence of powerful tools for studying neural circuitry, and the development of new approaches for exploring molecular mechanisms, interdisciplinary research on the pathogenesis of neurodevelopmental disorders is now increasingly common. Journal of Neurodevelopmental Disorders provides a unique venue for researchers interested in comparing and contrasting mechanisms and characteristics related to the pathogenesis of the full range of neurodevelopmental disorders, sharpening our understanding of the etiology and relevant phenotypes of each condition.
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