Xu Jinhong, Kong Yufei, Wang Nawen, Li Huijuan, Li Yunteng, Liu Zhuo, Yang Yuling, Yu Xiao, Liu Huihui, Ding Jing, Wang Yi, Zhao Rui, Shao Zhicheng
Institute for Translational Brain Research, State Key Laboratory of Medical Neurobiology, MOE Frontiers Center for Brain Science, Institute of Pediatrics, National Children's Medical Center, Children's Hospital, Fudan University, Shanghai, 200032, China.
Department of Neurology, Zhongshan Hospital, Fudan University, Shanghai, 200032, China.
Adv Sci (Weinh). 2025 Feb;12(8):e2409774. doi: 10.1002/advs.202409774. Epub 2024 Dec 31.
Focal cortical dysplasia (FCD) is a highly heterogeneous neurodevelopmental malformation, the underlying mechanisms of which remain largely elusive. In this study, personalized dorsal and ventral forebrain organoids (DFOs/VFOs) are generated derived from brain astrocytes of patients with FCD type II (FCD II). The pathological features of dysmorphic neurons, balloon cells, and astrogliosis are successfully replicated in patient-derived DFOs, but not in VFOs. It is noteworthy that cardiomyocyte-like cells correlated with dysmorphic neurons are generated through the high activation of BMP and WNT signaling in some of the FCD-organoids and patient cortical tissues. Moreover, functional assessments demonstrated the occurrence of epileptiform burst firing and propagative self-assembling neuronal hyperactivity in both FCD-DFOs and VFOs. Additionally, the heterotopic cardiomyocyte-organoids demonstrated the capacity for cardiomyocyte contraction and rhythmic firing. The presence of these cardiomyocytes contributes to the hyperactivity of neural networks in cardioids-DFOs assembly. In conclusion, the personalized region-specific forebrain organoids derived from FCD patient astrocytes effectively recapitulate heterogeneous pathological features, offering a valuable platform for the development of precise therapeutic strategies.
局灶性皮质发育不良(FCD)是一种高度异质性的神经发育畸形,其潜在机制在很大程度上仍不清楚。在本研究中,从II型FCD(FCD II)患者的脑星形胶质细胞中生成了个性化的背侧和腹侧前脑类器官(DFO/VFO)。在源自患者的DFO中成功复制了发育异常的神经元、气球样细胞和星形胶质细胞增生的病理特征,但在VFO中未复制。值得注意的是,在一些FCD类器官和患者皮质组织中,通过BMP和WNT信号的高度激活产生了与发育异常神经元相关的心肌样细胞。此外,功能评估表明,FCD-DFO和VFO中均出现了癫痫样爆发性放电和传播性自组装神经元活动亢进。此外,异位心肌类器官表现出心肌细胞收缩和节律性放电的能力。这些心肌细胞的存在导致了心形DFO组装中神经网络的活动亢进。总之,源自FCD患者星形胶质细胞的个性化区域特异性前脑类器官有效地概括了异质性病理特征,为开发精确的治疗策略提供了一个有价值的平台。