Li Dongyun, Feng Danyong, Hu Chunchun, Tian Yuxin, Reiss Allan L, Hagerman Randi J, Xu Xiu, Qiao Zhongwei, Li Rihui, Xu Qiong
Department of Child Health Care, Children's Hospital of Fudan University, Shanghai, China.
Centre for Cognitive and Brain Sciences, Institute of Collaborative Innovation, University of Macau, Taipa, Macau S.A.R., China.
Mol Psychiatry. 2025 Jul 15. doi: 10.1038/s41380-025-03112-y.
Autism spectrum disorder (ASD) and fragile X syndrome (FXS) are behaviorally overlapped. However, little is known about the functional patterns underlying the cognitive and behavioral characteristics of FXS and ASD. The present study aimed to identify the distinct or/and shared functional networks in young children with FXS and idiopathic ASD. We recruited 150 children consecutively in a group with FXS, a group with idiopathic ASD, and a group with typically developing (TD) children. Resting-state functional magnetic resonance imaging (fMRI) and behavioral data were collected and genetic information was obtained in the FXS group. We compared functional connectivity (FC) among the three groups and found that both FXS and ASD showed significantly decreased FC among the default mode network (DMN), sensorimotor network (SMN), cerebellum network (CN), and visual network (VN) relative to TD. FXS specifically demonstrated decreased FC within DMN, while both FXS and ASD exhibited significantly decreased FC within the CN and also between the CN and DMN, SMN, VN, respectively. Aberrant topological alterations of CN were identified in children with FXS and ASD, while ASD group showed significantly lower segregation in regions that integrate sensory and visual information, and motor coordination function. Moreover, correlations between the severity of social affect and mean FC of various cerebral-cerebellum networks in FXS exhibited significantly distinct trends from those observed in ASD. In the FXS group, the topological measure at crus I of the cerebellum is found to be negatively associated with DNA methylation levels. These results were statistically robust and demonstrated the shared and distinct profiles of intrinsic functional networks in FXS and ASD, two phenotypically overlapping developmental disorders.
自闭症谱系障碍(ASD)和脆性X综合征(FXS)在行为上存在重叠。然而,对于FXS和ASD认知及行为特征背后的功能模式,我们知之甚少。本研究旨在识别患有FXS和特发性ASD的幼儿中不同的或/和共享的功能网络。我们连续招募了150名儿童,分为FXS组、特发性ASD组和发育正常(TD)儿童组。收集了静息态功能磁共振成像(fMRI)数据和行为数据,并在FXS组中获取了基因信息。我们比较了三组之间的功能连接性(FC),发现相对于TD组,FXS和ASD在默认模式网络(DMN)、感觉运动网络(SMN)、小脑网络(CN)和视觉网络(VN)中的FC均显著降低。FXS在DMN内的FC尤其降低,而FXS和ASD在CN内以及CN与DMN、SMN、VN之间的FC均显著降低。在患有FXS和ASD的儿童中发现了CN的异常拓扑改变,而ASD组在整合感觉和视觉信息以及运动协调功能的区域中显示出显著更低的分离度。此外,FXS中社交情感严重程度与各种脑-小脑网络平均FC之间的相关性呈现出与ASD中观察到的显著不同的趋势。在FXS组中,发现小脑I脚的拓扑测量值与DNA甲基化水平呈负相关。这些结果在统计学上具有稳健性,证明了FXS和ASD这两种表型重叠的发育障碍在内在功能网络方面的共同和不同特征。