Division of Biomedical Sciences, School of Medicine, University of California, Riverside, CA, USA.
Department of Psychology, University of California Riverside, Riverside, CA, USA.
J Neurochem. 2020 Dec;155(5):538-558. doi: 10.1111/jnc.15037. Epub 2020 Jun 8.
Individuals with Fragile X Syndrome (FXS) and autism spectrum disorder (ASD) exhibit cognitive impairments, social deficits, increased anxiety, and sensory hyperexcitability. Previously, we showed that elevated levels of matrix metalloproteinase-9 (MMP-9) may contribute to abnormal development of parvalbumin (PV) interneurons and perineuronal nets (PNNs) in the developing auditory cortex (AC) of Fmr1 knock-out (KO) mice, which likely underlie auditory hypersensitivity. Thus, MMP-9 may serve as a potential target for treatment of auditory hypersensitivity in FXS. Here, we used the MMP-2/9 inhibitor, SB-3CT, to pharmacologically inhibit MMP-9 activity during a specific developmental period and to test whether inhibition of MMP-9 activity reverses neural oscillation deficits and behavioral impairments by enhancing PNN formation around PV cells in Fmr1 KO mice. Electroencephalography (EEG) was used to measure resting state and sound-evoked electrocortical activity in auditory and frontal cortices of postnatal day (P)22-23 male mice before and one-day after treatment with SB-3CT (25 mg/kg) or vehicle. At P27-28, animal behaviors were tested to measure the effects of the treatment on anxiety and hyperactivity. Results show that acute inhibition of MMP-9 activity improved evoked synchronization to auditory stimuli and ameliorated mouse behavioral deficits. MMP-9 inhibition enhanced PNN formation, increased PV levels and TrkB phosphorylation yet reduced Akt phosphorylation in the AC of Fmr1 KO mice. Our results show that MMP-9 inhibition during early postnatal development is beneficial in reducing some auditory processing deficits in the FXS mouse model and may serve as a candidate therapeutic for reversing sensory hypersensitivity in FXS and possibly other ASDs.
脆性 X 综合征(FXS)和自闭症谱系障碍(ASD)患者表现出认知障碍、社交缺陷、焦虑增加和感觉过度敏感。以前,我们发现基质金属蛋白酶-9(MMP-9)水平升高可能导致 Fmr1 敲除(KO)小鼠发育中的听觉皮层(AC)中的副钙蛋白(PV)中间神经元和周围神经网(PNN)异常发育,这可能是听觉过敏的基础。因此,MMP-9 可能成为 FXS 听觉过敏治疗的潜在靶点。在这里,我们使用 MMP-2/9 抑制剂 SB-3CT 在特定的发育时期抑制 MMP-9 活性,并通过增强 Fmr1 KO 小鼠中 PV 细胞周围的 PNN 形成来测试抑制 MMP-9 活性是否可以逆转神经振荡缺陷和行为障碍。脑电图(EEG)用于测量 P22-23 天雄性小鼠在接受 SB-3CT(25mg/kg)或载体处理前后的听觉和额叶皮层的静息状态和声音诱发的电皮质活动。在 P27-28,进行动物行为测试以测量处理对焦虑和多动的影响。结果表明,MMP-9 活性的急性抑制改善了对听觉刺激的诱发同步,并改善了小鼠的行为缺陷。MMP-9 抑制增强了 PNN 形成,增加了 PV 水平和 TrkB 磷酸化,但减少了 Fmr1 KO 小鼠 AC 中的 Akt 磷酸化。我们的结果表明,在出生后早期发育过程中抑制 MMP-9 活性有利于减少 FXS 小鼠模型中的一些听觉处理缺陷,并且可能作为逆转 FXS 及其他 ASD 中感觉过敏的候选治疗方法。