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儿童结节性硬化症患者静息态功能连接的改变。

Alterations in resting-state functional connectivity in pediatric patients with tuberous sclerosis complex.

机构信息

Department of Neurology, Washington University, St. Louis, MO, USA.

Department of Radiology, Washington University, St. Louis, MO, USA.

出版信息

Epilepsia Open. 2021 Sep;6(3):579-587. doi: 10.1002/epi4.12523. Epub 2021 Aug 3.

Abstract

OBJECTIVE

To investigate resting-state functional connectivity (FC) in pediatric patients with tuberous sclerosis complex and intractable epilepsy requiring surgery.

METHODS

Resting-state functional MRI was utilized to investigate functional connectivity in 13 pediatric patients with tuberous sclerosis complex (TSC) and intractable epilepsy requiring surgery.

RESULTS

The majority of patients demonstrated a resting-state network architecture similar to those reported in healthy individuals. However, preoperative differences were evident between patients with high versus low tuber burden, as well as those with good versus poor neurodevelopmental outcomes, most notably in the cingulo-opercular and visual resting-state networks. One patient with high tuber burden and poor preoperative development and seizure control had nearly normal development and seizure resolution after surgery. This was accompanied by significant improvement in resting-state network architecture just one day postoperatively.

SIGNIFICANCE

Although many patients with tuberous sclerosis complex and medically refractory epilepsy demonstrate functional connectivity patterns similar to healthy children, relationships within and between RSNs demonstrate clear differences in patients with higher tuber burden and worse outcomes. Improvements in resting-state network organization postoperatively may be related to epilepsy surgery outcomes, providing candidate biomarkers for clinical management in this high-risk population.

摘要

目的

研究需要手术治疗的结节性硬化症伴耐药性癫痫儿童的静息态功能连接。

方法

利用静息态功能磁共振成像研究了 13 名患有结节性硬化症且需要手术治疗的耐药性癫痫的儿科患者的功能连接。

结果

大多数患者表现出与健康个体相似的静息态网络结构。然而,术前高与低结节负荷的患者之间以及神经发育结局好与差的患者之间存在差异,尤其是在扣带回-脑岛和视觉静息态网络中。一名高结节负荷、术前发育和癫痫控制不良的患者术后几乎正常发育,癫痫得到缓解。术后仅一天,静息态网络结构就显著改善。

意义

尽管许多患有结节性硬化症和药物难治性癫痫的儿童表现出与健康儿童相似的功能连接模式,但 RSN 内和 RSN 之间的关系在结节负荷较高和预后较差的患者中表现出明显差异。术后静息态网络组织的改善可能与癫痫手术结果有关,为该高危人群的临床管理提供了候选生物标志物。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/247b/8408601/c69f0b8e256e/EPI4-6-579-g002.jpg

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