Samuelsson Kristin, Osman Ayman A M, Angeria Maria, Risling Mårten, Mohseni Simin, Press Rayomand
Department of Clinical Neuroscience, Department of Neurology, Karolinska Institutet, Karolinska University Hospital, Stockholm, Sweden.
Department of Clinical and Experimental Medicine, Division of Cell Biology, Linköping University, Linköping, Sweden.
PLoS One. 2016 Sep 23;11(9):e0163427. doi: 10.1371/journal.pone.0163427. eCollection 2016.
Twenty-five percent of polyneuropathies are idiopathic. Microangiopathy has been suggested to be a possible pathogenic cause of chronic idiopathic axonal polyneuropathy (CIAP). Dysfunction of the autophagy pathway has been implicated as a marker of neurodegeneration in the central nervous system, but the autophagy process is not explored in the peripheral nervous system. In the current study, we examined the presence of microangiopathy and autophagy-related structures in sural nerve biopsies of 10 patients with CIAP, 11 controls with inflammatory neuropathy and 10 controls without sensory polyneuropathy. We did not find any significant difference in endoneurial microangiopathic markers in patients with CIAP compared to normal controls, though we did find a correlation between basal lamina area thickness and age. Unexpectedly, we found a significantly larger basal lamina area thickness in patients with vasculitic neuropathy. Furthermore, we found a significantly higher density of endoneurial autophagy-related structures, particularly in patients with CIAP but also in patients with inflammatory neuropathy, compared to normal controls. It is unclear if the alteration in the autophagy pathway is a consequence or a cause of the neuropathy. Our results do not support the hypothesis that CIAP is primarily caused by a microangiopathic process in endoneurial blood vessels in peripheral nerves. The significantly higher density of autophagy structures in sural nerves obtained from patients with CIAP and inflammatory neuropathy vs. controls indicates the involvement of this pathway in neuropathy, particularly in CIAP, since the increase in density of autophagy-related structures was more pronounced in patients with CIAP than those with inflammatory neuropathy. To our knowledge this is the first report investigating signs of autophagy process in peripheral nerves in patients with CIAP and inflammatory neuropathy.
25%的多发性神经病为特发性。微血管病变被认为可能是慢性特发性轴索性多发性神经病(CIAP)的致病原因。自噬途径功能障碍被认为是中枢神经系统神经退行性变的一个标志物,但在外周神经系统中尚未对自噬过程进行研究。在本研究中,我们检查了10例CIAP患者、11例炎性神经病对照者和10例无感觉性多发性神经病对照者的腓肠神经活检组织中微血管病变和自噬相关结构的情况。与正常对照相比,我们未发现CIAP患者的神经内膜微血管病变标志物有任何显著差异,不过我们确实发现基膜面积厚度与年龄之间存在相关性。出乎意料的是,我们发现血管炎性神经病患者的基膜面积厚度显著更大。此外,与正常对照相比,我们发现神经内膜自噬相关结构的密度显著更高,特别是在CIAP患者中,炎性神经病患者中也如此。尚不清楚自噬途径的改变是神经病的结果还是原因。我们的结果不支持CIAP主要由外周神经神经内膜血管中的微血管病变过程引起这一假说。CIAP和炎性神经病患者与对照者相比,腓肠神经中自噬结构的密度显著更高,这表明该途径参与了神经病,特别是在CIAP中,因为自噬相关结构密度的增加在CIAP患者中比炎性神经病患者中更明显。据我们所知,这是第一份研究CIAP和炎性神经病患者外周神经自噬过程迹象的报告。