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Mitochondrial dysfunction triggers secretion of the immunosuppressive factor α-fetoprotein

Jett, K. A.; Baker, Z. N.; Hossain, A.; Boulet, A.; Cobine, P. A.; Ghosh, S.; Ng, P.; Yilmaz, O.; Barretto, K.; DeCoteau, J.; Mochoruk, K.; Ioannou, G.; Savard, C.; Yuan, S.; Lowden, C.; Kim, B.-E.; Cheng, H.-Y. M.; Battersby, B. J.; Gohil, V. M. M.; Leary, S. C.

2021-07-03 cell biology
10.1101/2021.07.02.450924 bioRxiv
Show abstract

Signaling circuits crucial to systemic physiology are widespread, yet uncovering their molecular underpinnings remains a barrier to understanding the etiology of many metabolic disorders. Here, we identify a copper-linked signaling circuit activated by disruption of mitochondrial function in the murine liver or heart that results in atrophy of the spleen and thymus and causes a peripheral white blood cell deficiency. We demonstrate that the leukopenia is caused by -fetoprotein, which requires copper and the cell surface receptor CCR5 to promote white blood cell death. We further show that -fetoprotein expression is upregulated in several cell types upon inhibition of oxidative phosphorylation, including a muscle cell model of Barth syndrome. Collectively, our data argue that -fetoprotein secreted by bioenergetically stressed tissue suppresses the immune system, an effect which may explain the recurrent infections that are observed in a subset of mitochondrial diseases or in other disorders with mitochondrial involvement.

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