Division of Biomedical Sciences, University of California Riverside School of Medicine, Riverside, CA, USA.
Neuroscience Graduate Program, University of California Riverside, Riverside, CA, USA.
Cereb Cortex. 2018 Nov 1;28(11):3951-3964. doi: 10.1093/cercor/bhx258.
Abnormal sensory responses associated with Fragile X Syndrome (FXS) and autism spectrum disorders include hypersensitivity and impaired habituation to repeated stimuli. Similar sensory deficits are also observed in adult Fmr1 knock-out (KO) mice and are reversed by genetic deletion of Matrix Metalloproteinase-9 (MMP-9) through yet unknown mechanisms. Here we present new evidence that impaired development of parvalbumin (PV)-expressing inhibitory interneurons may underlie hyper-responsiveness in auditory cortex of Fmr1 KO mice via MMP-9-dependent regulation of perineuronal nets (PNNs). First, we found that PV cell development and PNN formation around GABAergic interneurons were impaired in developing auditory cortex of Fmr1 KO mice. Second, MMP-9 levels were elevated in P12-P18 auditory cortex of Fmr1 KO mice and genetic reduction of MMP-9 to WT levels restored the formation of PNNs around PV cells. Third, in vivo single-unit recordings from auditory cortex neurons showed enhanced spontaneous and sound-driven responses in developing Fmr1 KO mice, which were normalized following genetic reduction of MMP-9. These findings indicate that elevated MMP-9 levels contribute to the development of sensory hypersensitivity by influencing formation of PNNs around PV interneurons suggesting MMP-9 as a new therapeutic target to reduce sensory deficits in FXS and potentially other autism spectrum disorders.
脆性 X 综合征 (FXS) 和自闭症谱系障碍相关的异常感觉反应包括对重复刺激的超敏反应和习惯化受损。成年 Fmr1 敲除 (KO) 小鼠也观察到类似的感觉缺陷,并且通过基质金属蛋白酶-9 (MMP-9) 的遗传缺失通过未知机制得到逆转。在这里,我们提出了新的证据,表明抑制性中间神经元表达的 Parvalbumin (PV) 发育障碍可能通过 MMP-9 依赖的 Perineuronal Nets (PNNs) 调节,导致 Fmr1 KO 小鼠听觉皮层的超敏反应。首先,我们发现 Fmr1 KO 小鼠发育中的听觉皮层中 PV 细胞的发育和 GABA 能中间神经元周围的 PNN 形成受损。其次,MMP-9 水平在 P12-P18 的 Fmr1 KO 小鼠听觉皮层中升高,并且 MMP-9 的遗传减少到 WT 水平恢复了 PV 细胞周围 PNN 的形成。第三,来自听觉皮层神经元的体内单细胞记录显示,在发育中的 Fmr1 KO 小鼠中自发和声音驱动的反应增强,在 MMP-9 的遗传减少后恢复正常。这些发现表明,升高的 MMP-9 水平通过影响 PV 中间神经元周围 PNN 的形成,导致感觉超敏反应的发展,提示 MMP-9 是减少 FXS 及潜在其他自闭症谱系障碍感觉缺陷的新治疗靶点。