Literature DB >> 18077684

Autonomic denervation of lymphoid organs leads to epigenetic immune atrophy in a mouse model of Krabbe disease.

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.

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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


  32 in total

1.  Factors that affect postnatal bone growth retardation in the twitcher murine model of Krabbe disease.

Authors:  Miguel Agustin Contreras; William Louis Ries; Srinivasan Shanmugarajan; Gonzalo Arboleda; Inderjit Singh; Avtar Kaur Singh
Journal:  Biochim Biophys Acta       Date:  2010-05-02

Review 2.  Epigenetic principles and mechanisms underlying nervous system functions in health and disease.

Authors:  Mark F Mehler
Journal:  Prog Neurobiol       Date:  2008-10-17       Impact factor: 11.685

3.  Mechanism-based combination treatment dramatically increases therapeutic efficacy in murine globoid cell leukodystrophy.

Authors:  Jacqueline A Hawkins-Salsbury; Lauren Shea; Xuntian Jiang; Daniel A Hunter; A Miguel Guzman; Adarsh S Reddy; Elizabeth Y Qin; Yedda Li; Steven J Gray; Daniel S Ory; Mark S Sands
Journal:  J Neurosci       Date:  2015-04-22       Impact factor: 6.167

Review 4.  Biochemical, cell biological, pathological, and therapeutic aspects of Krabbe's disease.

Authors:  Je-Seong Won; Avtar K Singh; Inderjit Singh
Journal:  J Neurosci Res       Date:  2016-11       Impact factor: 4.164

5.  Axonopathy is a compounding factor in the pathogenesis of Krabbe disease.

Authors:  Ludovico Cantuti Castelvetri; Maria Irene Givogri; Hongling Zhu; Benjamin Smith; Aurora Lopez-Rosas; Xi Qiu; Richard van Breemen; Ernesto Roque Bongarzone
Journal:  Acta Neuropathol       Date:  2011-03-04       Impact factor: 17.088

6.  Thymic involution and corticosterone level in Sandhoff disease model mice: new aspects the pathogenesis of GM2 gangliosidosis.

Authors:  Kazuhiko Matsuoka; Daisuke Tsuji; Takao Taki; Kohji Itoh
Journal:  J Inherit Metab Dis       Date:  2011-05-20       Impact factor: 4.982

7.  Psychosine inhibits osteoclastogenesis and bone resorption via G protein-coupled receptor 65.

Authors:  S H Ahn; S-Y Lee; J-E Baek; S-Y Lee; S-Y Park; Y-S Lee; H Kim; B-J Kim; S H Lee; J-M Koh
Journal:  J Endocrinol Invest       Date:  2015-04-05       Impact factor: 4.256

8.  Mechanism of neuromuscular dysfunction in Krabbe disease.

Authors:  Ludovico Cantuti-Castelvetri; Erick Maravilla; Michael Marshall; Tammy Tamayo; Ludovic D'auria; John Monge; James Jeffries; Tuba Sural-Fehr; Aurora Lopez-Rosas; Guannan Li; Kelly Garcia; Richard van Breemen; Charles Vite; Jesus Garcia; Ernesto R Bongarzone
Journal:  J Neurosci       Date:  2015-01-28       Impact factor: 6.167

Review 9.  Neural pathways involved in infection-induced inflammation: recent insights and clinical implications.

Authors:  Marion Griton; Jan Pieter Konsman
Journal:  Clin Auton Res       Date:  2018-03-14       Impact factor: 4.435

10.  Thymic alterations in GM2 gangliosidoses model mice.

Authors:  Seiichi Kanzaki; Akira Yamaguchi; Kayoko Yamaguchi; Yoshitsugu Kojima; Kyoko Suzuki; Noriko Koumitsu; Yoji Nagashima; Kiyotaka Nagahama; Michiko Ehara; Yoshio Hirayasu; Akihide Ryo; Ichiro Aoki; Shoji Yamanaka
Journal:  PLoS One       Date:  2010-08-10       Impact factor: 3.240

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