Iron overload causes progressive organ damage and is associated with arthritis, liver damage, and heart failure. Elevated iron levels are present in 1%-5% of individuals; however, iron overload is undermonitored and underdiagnosed. Genetic factors affecting iron homeostasis are emerging. Individuals with hereditary xerocytosis, a rare disorder with gain-of-function (GOF) mutations in mechanosensitive PIEZO1 ion channel, develop age-onset iron overload. We show that constitutive or macrophage expression of a GOF Piezo1 allele in mice disrupts levels of the iron regulator hepcidin and causes iron overload. We further show that PIEZO1 is a key regulator of macrophage phagocytic activity and subsequent erythrocyte turnover. Strikingly, we find that E756del, a mild GOF PIEZO1 allele present in one-third of individuals of African descent, is strongly associated with increased plasma iron. Our study links macrophage mechanotransduction to iron metabolism and identifies a genetic risk factor for increased iron levels in African Americans.

A role of PIEZO1 in iron metabolism in mice and humans / Ma, S.; Dubin, A. E.; Zhang, Y.; Mousavi, S. A. R.; Wang, Y.; Coombs, A. M.; Loud, M.; Andolfo, I.; Patapoutian, A.. - In: CELL. - ISSN 0092-8674. - 184:4(2021), pp. 969-982.e13. [10.1016/j.cell.2021.01.024]

A role of PIEZO1 in iron metabolism in mice and humans

Andolfo I.
Conceptualization
;
2021

Abstract

Iron overload causes progressive organ damage and is associated with arthritis, liver damage, and heart failure. Elevated iron levels are present in 1%-5% of individuals; however, iron overload is undermonitored and underdiagnosed. Genetic factors affecting iron homeostasis are emerging. Individuals with hereditary xerocytosis, a rare disorder with gain-of-function (GOF) mutations in mechanosensitive PIEZO1 ion channel, develop age-onset iron overload. We show that constitutive or macrophage expression of a GOF Piezo1 allele in mice disrupts levels of the iron regulator hepcidin and causes iron overload. We further show that PIEZO1 is a key regulator of macrophage phagocytic activity and subsequent erythrocyte turnover. Strikingly, we find that E756del, a mild GOF PIEZO1 allele present in one-third of individuals of African descent, is strongly associated with increased plasma iron. Our study links macrophage mechanotransduction to iron metabolism and identifies a genetic risk factor for increased iron levels in African Americans.
2021
A role of PIEZO1 in iron metabolism in mice and humans / Ma, S.; Dubin, A. E.; Zhang, Y.; Mousavi, S. A. R.; Wang, Y.; Coombs, A. M.; Loud, M.; Andolfo, I.; Patapoutian, A.. - In: CELL. - ISSN 0092-8674. - 184:4(2021), pp. 969-982.e13. [10.1016/j.cell.2021.01.024]
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Utilizza questo identificativo per citare o creare un link a questo documento: https://hdl.handle.net/11588/866894
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