Novel unconventional myosins in B cell homeostasis

NIH RePORTER · NIH · R01 · $529,302 · view on reporter.nih.gov ↗

Abstract

ABSTRACT Incurable autoimmune diseases afflict millions of Americans, cause pain and persistent damage to multiple organ systems, and have a major impact on patient health, well-being and quality of life. Increased B cell numbers, greater B cell activation and differentiation into plasma cells, and autoantibody production often characterize autoimmunity, signifying that dysregulated B cell survival and differentiation are critical steps in autoimmune pathogenesis. Identification of proteins and molecular events that regulate B cell survival, activation and differentiation is therefore critical for better mechanistic understanding of autoimmune disease development. Myo18A is a scaffolding protein with a unique domain organization that enables assembly of multiprotein complexes in various subcellular compartments of non-immune cells. We reported that B cells express Myo18A, and that its conditional deletion in B cells (Myo18A BKO) leads to an increase in mature B cells and plasma cells, splenomegaly, hypergammaglobulinemia, and development of autoantibodies. Interestingly, this profile overlaps with autoimmune manifestations reported in transgenic mice overexpressing the B cell survival factor BAFF, and in mice with genetic deletion of proteins involved in mRNA decay. In preliminary data, we observed that Myo18A-deficient B cells display features of greater BAFF responsiveness, including increased mitochondrial size and gene expression, and stronger pro-survival Akt signaling, indicating that Myo18A negatively regulates B cell response to BAFF. Additionally, deletion of Myo18A in B cells increased the basal expression and reduced the decay of mRNA encoding Blimp-1, a key transcription factor in B cell differentiation. Further, Myo18A in B cells bound to Blimp1 mRNA and co-localized with subcellular mRNA degradation sites, indicating that Myo18A is involved in post-transcriptional regulation and expression of Blimp- 1. Our published and preliminary data suggest that Myo18A is an important and previously unrecognized determinant in B cell immunity. Our specific aims are to test the hypotheses that (1) Myo18A restricts BCR and BAFF-R signaling to control B cell homeostasis, (2) Myo18A inhibits mRNA stability to inform B cell gene expression, and (3) Myo18A limits B cell differentiation by controlling the B cell transcriptome. The proposed research is innovative because it will establish a novel role for the unconventional myosin family protein Myo18A in preventing uncontrolled B cell activation and differentiation. Specifically, our research will identify a novel functional connection between Myo18A and B cell survival and differentiation through regulation of B cell antigen and BAFF responsiveness and post-transcriptional mRNA stability. This research is expected to have significant impact because it will enable better mechanistic understanding of the molecular processes that prevent exaggerated antibody development.

Key facts

NIH application ID
10848359
Project number
5R01AI172916-02
Recipient
CLEVELAND CLINIC LERNER COM-CWRU
Principal Investigator
Neetu Gupta
Activity code
R01
Funding institute
NIH
Fiscal year
2024
Award amount
$529,302
Award type
5
Project period
2023-06-01 → 2028-05-31