It is suggested that the Srsf2 P95H/WT mutation contributes to the altered hematopoietic lineages composition and immune-related cellular states in BM, providing insights into its potential involvement in early-stage MDS and CMML progression.
Abstract
Somatic
SRSF2
mutations are early lesions in myelodysplastic syndromes (MDS) and chronic myelomonocytic leukemia (CMML), yet how they contribute disease remain unclear. In this study, we generated two murine models that have not yet developed overt MDS/CMML-like phenotypes: (i) a cell-derived xenograft (CDX) by tail-vein injection of stable BA/F3-
KRAS
G12C
-
SRSF2
P95H
cells and (ii) a Vav1-Cre conditional knock-in mouse heterozygous for
Srsf2
P95H
. Molecular assays, bulk RNA-seq, and single-cell RNA-seq (scRNA-seq) were used to profile the bone-marrow (BM) microenvironment. CDX mice carrying
SRSF2
P95H
displayed systemic immune dysregulation. Bulk RNA-seq of Vav1-Cre BM revealed attenuated T cell chemotaxis and elevated immunoglobulin production in
Srsf2
P95H/WT
animals. Extensive mis-splicing of spliceosomal, epigenetic, and cytoskeletal regulators (
Hnrnpa2b1, Rsrp1, H2az1, Erbin, Akap13
) was also detected. scRNA-seq demonstrated a reduction in hematopoietic stem and progenitor cells (HSPCs) and an expansion of neutrophil precursors in
Srsf2
P95H/WT
mice, verifying by flow-cytometry assay. A loss of Natural killer (NK) cells and effector T cells, together with a shift of the B-cell lineage from early precursor states toward more mature and transcriptionally active populations were detected in
Srsf2
P95H/WT
group, accompanied by enrichment of antibody secretion–related programs. Sanger sequencing confirmed the presence of Srsf2 mutations in this B-cell subset and showed altered expression and alternative splicing of
Hnrnpa2b1
, a well-studied target of SRSF2 with P95 mutation. Collectively, these findings suggest that the
Srsf2
P95H/WT
mutation contributes to the altered hematopoietic lineages composition and immune-related cellular states in BM, providing insights into its potential involvement in early-stage MDS and CMML progression.
It is demonstrated that SLF2 and SMC5 dysfunction drives premature HSC aging, bone marrow failure, and predisposition to MDS, revealing Atelis Syndrome as a previously unrecognized IBMFS.
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