Max Planck Institute for Human Cognitive and Brain Sciences, Leipzig, Germany; Department of Psychiatry, Social Psychiatry and Psychotherapy, Hannover Medical School, Hannover, Germany.
Center for Human Genetics Research, Massachusetts General Hospital, Boston, MA, USA; Harvard Medical School, Boston, MA, USA.
Neurobiol Dis. 2023 Sep;185:106252. doi: 10.1016/j.nbd.2023.106252. Epub 2023 Aug 2.
Gilles de la Tourette syndrome (GTS) is a neuropsychiatric movement disorder with reported abnormalities in various neurotransmitter systems. Considering the integral role of iron in neurotransmitter synthesis and transport, it is hypothesized that iron exhibits a role in GTS pathophysiology. As a surrogate measure of brain iron, quantitative susceptibility mapping (QSM) was performed in 28 patients with GTS and 26 matched controls. Significant susceptibility reductions in the patients, consistent with reduced local iron content, were obtained in subcortical regions known to be implicated in GTS. Regression analysis revealed a significant negative association of tic scores and striatal susceptibility. To interrogate genetic mechanisms that may drive these reductions, spatially specific relationships between susceptibility and gene-expression patterns from the Allen Human Brain Atlas were assessed. Correlations in the striatum were enriched for excitatory, inhibitory, and modulatory neurochemical signaling mechanisms in the motor regions, mitochondrial processes driving ATP production and iron‑sulfur cluster biogenesis in the executive subdivision, and phosphorylation-related mechanisms affecting receptor expression and long-term potentiation in the limbic subdivision. This link between susceptibility reductions and normative transcriptional profiles suggests that disruptions in iron regulatory mechanisms are involved in GTS pathophysiology and may lead to pervasive abnormalities in mechanisms regulated by iron-containing enzymes.
图雷特综合征(Gilles de la Tourette syndrome,GTS)是一种神经精神运动障碍,据报道其涉及多种神经递质系统的异常。鉴于铁在神经递质合成和转运中的重要作用,因此假设铁在 GTS 病理生理学中发挥作用。作为脑铁的替代测量方法,对 28 名 GTS 患者和 26 名匹配对照进行了定量磁敏感图(quantitative susceptibility mapping,QSM)。在已知与 GTS 相关的皮质下区域获得了患者的显著磁敏感降低,与局部铁含量降低一致。回归分析显示, tic 评分与纹状体的磁敏感呈显著负相关。为了探究可能导致这些减少的遗传机制,评估了 Allen 人类大脑图谱中基因表达模式与易感性之间的空间特异性关系。纹状体中的相关性在运动区域中富含兴奋性、抑制性和调节性神经化学信号机制,在执行子区域中富含驱动 ATP 产生和铁硫簇生物发生的线粒体过程,以及在边缘子区域中影响受体表达和长时程增强的磷酸化相关机制。易感性降低与规范转录谱之间的这种联系表明,铁调节机制的破坏与 GTS 病理生理学有关,并可能导致含铁酶调节的机制普遍异常。