Krull Frederik, Hosseini Shahrbanou, Bleyer Martina, Brenig Bertram
Div. of Molecular Biology of Livestock and Molecular Diagnostics, Georg-August University Göttingen, Institute of Veterinary Medicine, Göttingen, Germany.
German Primate Center, Pathology Unit, Leibniz Institute for Primate Research, Göttingen, Germany.
PLoS One. 2025 May 29;20(5):e0324633. doi: 10.1371/journal.pone.0324633. eCollection 2025.
Bovine spastic paresis (BSP) is a progressive neuromuscular disease of unknown origin that causes persistent stiffness of the hind limbs. The symptoms are similar to those of human motor neuron diseases such as primary (PLS) or amyotrophic lateral sclerosis (ALS). BSP occurs worldwide in cattle production with an estimated prevalence of <1%. For Germany, this means that around 20,000 Holstein cattle are affected. BSP is generally considered a hereditary disease, but there is no prevention through breeding programs. As a result, BSP not only affects animal welfare but also leads to economic losses in milk and beef production. Here, we used transcriptomics to analyse the brainstem, spinal cord and affected gastrocnemius muscle tissue of eight animals affected by BSP and eight control animals from slaughterhouses to gain new insights into the molecular mechanisms underlying BSP. We found that the expression of several genes was significantly different in animals affected by BSP compared to control animals. Specific genes for inhibitory neurons were downregulated in the brainstems of the affected animals, namely CCK (cholecystokinin), NPY (neuropeptide Y), and SST (somatostatin). These inhibitory neurotransmitters influence cerebral movement control, among other processes. Furthermore, OOSP2 (oocyte secreted protein 2) was found to be significantly upregulated in the affected animals in all tissues. This expression could best be explained by the presence of T-follicular-helper cells which, through interleukin 21, can trigger a TH-2-dominated immune response and lead to autoimmune encephalitis. Further cases were sampled for confirmation and we detected cell infiltrates of activated microglia and T-cells in the brainstem using immunohistochemistry. Microglial foci were significantly more abundant in animals affected by BSP than control animals. We conclude that BSP is caused by an autoimmune reaction directed against inhibitory interneurons in the brainstem and is due to a combination of genetics and environmental influences. This may result in lost controlling influence on the upper motor neurons via extrapyramidal pathways and therefore triggers the specific symptoms of motor neuron disease.
牛痉挛性轻瘫(BSP)是一种病因不明的进行性神经肌肉疾病,可导致后肢持续僵硬。其症状与人类运动神经元疾病如原发性侧索硬化症(PLS)或肌萎缩侧索硬化症(ALS)相似。BSP在全球范围内的养牛业中都有发生,估计患病率<1%。就德国而言,这意味着约20000头荷斯坦奶牛受到影响。BSP通常被认为是一种遗传性疾病,但通过育种计划无法预防。因此,BSP不仅影响动物福利,还会导致牛奶和牛肉生产的经济损失。在此,我们利用转录组学分析了8头受BSP影响的动物和8头来自屠宰场的对照动物的脑干、脊髓及受影响的腓肠肌组织,以深入了解BSP潜在的分子机制。我们发现,与对照动物相比,受BSP影响的动物中几个基因的表达存在显著差异。受影响动物脑干中抑制性神经元的特定基因表达下调,即胆囊收缩素(CCK)、神经肽Y(NPY)和生长抑素(SST)。这些抑制性神经递质在其他过程中影响大脑运动控制。此外,在所有组织中,受影响动物的卵母细胞分泌蛋白2(OOSP2)被发现显著上调。这种表达最合理的解释是存在T滤泡辅助细胞,其可通过白细胞介素21引发以TH-2为主导的免疫反应并导致自身免疫性脑炎。采集更多病例进行确认,我们通过免疫组织化学检测到脑干中有活化的小胶质细胞和T细胞浸润。受BSP影响的动物中的小胶质细胞病灶明显比对照动物丰富。我们得出结论,BSP是由针对脑干中抑制性中间神经元的自身免疫反应引起的,是遗传和环境影响共同作用的结果。这可能导致通过锥体外系途径对上运动神经元的控制影响丧失,从而引发运动神经元疾病的特定症状。