| Literature DB >> 18077684 |
Francesca Galbiati1, Veronica Basso, Ludovico Cantuti, Maria Irene Givogri, Aurora Lopez-Rosas, Nicolas Perez, Chenthamarakshan Vasu, Hongmei Cao, Richard van Breemen, Anna Mondino, Ernesto R Bongarzone.
Abstract
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.Entities:
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Year: 2007 PMID: 18077684 PMCID: PMC6673629 DOI: 10.1523/JNEUROSCI.3379-07.2007
Source DB: PubMed Journal: J Neurosci ISSN: 0270-6474 Impact factor: 6.167