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JMJD6 promotes self-renewal and regenerative capacity of hematopoietic stem cells.

Published version
Peer-reviewed

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Authors

Lawson, Hannah 
Sepulveda, Catarina 
van de Lagemaat, Louie N 
Durko, Jozef 
Barile, Melania 

Abstract

Lifelong multilineage hematopoiesis critically depends on rare hematopoietic stem cells (HSCs) that reside in the hypoxic bone marrow microenvironment. Although the role of the canonical oxygen sensor hypoxia-inducible factor prolyl hydroxylase has been investigated extensively in hematopoiesis, the functional significance of other members of the 2-oxoglutarate (2-OG)-dependent protein hydroxylase family of enzymes remains poorly defined in HSC biology and multilineage hematopoiesis. Here, by using hematopoietic-specific conditional gene deletion, we reveal that the 2-OG-dependent protein hydroxylase JMJD6 is essential for short- and long-term maintenance of the HSC pool and multilineage hematopoiesis. Additionally, upon hematopoietic injury, Jmjd6-deficient HSCs display a striking failure to expand and regenerate the hematopoietic system. Moreover, HSCs lacking Jmjd6 lose multilineage reconstitution potential and self-renewal capacity upon serial transplantation. At the molecular level, we found that JMJD6 functions to repress multiple processes whose downregulation is essential for HSC integrity, including mitochondrial oxidative phosphorylation (OXPHOS), protein synthesis, p53 stabilization, cell cycle checkpoint progression, and mTORC1 signaling. Indeed, Jmjd6-deficient primitive hematopoietic cells display elevated basal and maximal mitochondrial respiration rates and increased reactive oxygen species (ROS), prerequisites for HSC failure. Notably, an antioxidant, N-acetyl-l-cysteine, rescued HSC and lymphoid progenitor cell depletion, indicating a causal impact of OXPHOS-mediated ROS generation upon Jmjd6 deletion. Thus, JMJD6 promotes HSC maintenance and multilineage differentiation potential by suppressing fundamental pathways whose activation is detrimental for HSC function.

Description

Keywords

Bone Marrow, Bone Marrow Transplantation, Cell Differentiation, Hematopoiesis, Hematopoietic Stem Cells

Journal Title

Blood Adv

Conference Name

Journal ISSN

2473-9529
2473-9537

Volume Title

5

Publisher

American Society of Hematology

Rights

All rights reserved
Sponsorship
Medical Research Council (MC_PC_17230)
This work is funded by a project grant from Blood Cancer UK (formerly Bloodwise). K.R.K.’s laboratory is also supported by Cancer Research UK Programme Grant, The Barts Charity, Medical Research Council, and the Kay Kendall Leukaemia Fund. N.M.M. was funded by a NIA from Wellcome. A.L. and D.V. laboratory received support from the BBSRC Institute Strategic Program Funding