Galbiati Francesca, Basso Veronica, Cantuti Ludovico, Givogri Maria Irene, Lopez-Rosas Aurora, Perez Nicolas, Vasu Chenthamarakshan, Cao Hongmei, van Breemen Richard, Mondino Anna, Bongarzone Ernesto R
Department of Anatomy, College of Medicine, University of Illinois at Chicago, Chicago, Illinois 60612-7308, USA.
J Neurosci. 2007 Dec 12;27(50):13730-8. doi: 10.1523/JNEUROSCI.3379-07.2007.
Lysosomal beta-galactosylceramidase deficiency results in demyelination and inflammation in the nervous system causing the neurological Krabbe disease. In the Twitcher mouse model of this disease, we found that neurological symptoms parallel progressive and severe lymphopenia. Although lymphopoiesis is normal before disease onset, primary and secondary lymphoid organs progressively degenerate afterward. This occurs despite preserved erythropoiesis and leads to severe peripheral lymphopenia caused by reduced numbers of T cell precursors and mature lymphocytes. Hematopoietic cell replacement experiments support the existence of an epigenetic factor in mutant mice reconcilable with a progressive loss of autonomic axons that hampers thymic functionality. We propose that degeneration of autonomic nerves leads to the irreversible thymic atrophy and loss of immune-competence. Our study describes a new aspect of Krabbe disease, placing patients at risk of immune-related pathologies, and identifies a novel target for therapeutic interventions.
溶酶体β-半乳糖神经酰胺酶缺乏会导致神经系统脱髓鞘和炎症,引发神经学上的克拉伯病。在该疾病的抽搐小鼠模型中,我们发现神经学症状与进行性严重淋巴细胞减少平行。尽管在疾病发作前淋巴细胞生成正常,但原发性和继发性淋巴器官随后会逐渐退化。尽管红细胞生成得以保留,但仍会发生这种情况,并导致由T细胞前体和成熟淋巴细胞数量减少引起的严重外周淋巴细胞减少。造血细胞替代实验支持突变小鼠中存在一种表观遗传因素,这与妨碍胸腺功能的自主神经轴突的渐进性丧失相一致。我们提出自主神经变性会导致不可逆的胸腺萎缩和免疫能力丧失。我们的研究描述了克拉伯病的一个新方面,使患者面临免疫相关病理的风险,并确定了一个新的治疗干预靶点。