Canonical nuclear envelope protein emerin regulates structure and integrity of the erythrocyte plasma membrane
Brendan O'BrieniD, Joyleen Oliver, Evrett Thompson, Madeline Y. MaydayiD, Rubia Isler MancusoiD, Alokkhumar Jha, Jennifer M. KwaniD, Gisèle BonneiD, Rabah Ben YaouiD, France LeturcqiD, Suxia BaiiD, Timothy NottoliiD, John HwaiD, Diane S. KrauseiD, Patrick G. GallagheriD, Sean X. GuiD, Kathleen A. MartiniD
Abstract
Red blood cells (RBC) are essential for the survival of aerobic organisms. Mutations in RBC plasma membrane proteins can give rise to a spectrum of diseases characterized by hemolysis, inefficient gas exchange, and anemia. We tested whether emerin (EMD), a nuclear envelope (NE) protein, is also expressed in anuclear cell types to regulate membrane structure and function. We find that emerin is localized to the RBC plasma membrane and regulates the localization of other membrane proteins involved in actin branching and cytoskeletal dynamics. Additionally, we find that loss of Emd in mice results in decreased RBC corpuscular volume and hemoglobin content in both sexes, as well as a male-specific increase in RBC number and hematocrit, consistent with other male-dominated EMD phenotypes. Along with decreased RBC size, we observed a reduction in the rate of osmotic lysis in Emd KO animals, particularly males. We found that human EMD mutations also have a statistically significant association with RBC phenotypes, namely MCV. Individuals with emerin-related Emery-Dreifuss muscular dystrophy (EDMD) also demonstrate a mild yet consistent RBC phenotype according to patient CBC data. Together, these data suggest that emerin can have cell-specific localization and functions independent of its canonical repertoire in nucleated cells.
02 /Specification & Classification
Click any tag to browse matching papers- Citations
- 0
- Organism
- Assay
- Research Focus
- Approach
Reproducibility ledger
Nobody has reported trying to reproduce this paper yet. Did you replicate an experiment or run the code? Your report saves the next lab a week.
Sign in to file a reproducibility report.
Related papers
- The inner nuclear membrane protein NEMP1 is required for nuclear envelope openings and enucleation of erythroblasts during erythropoiesis
D. Hodzic, J. Wu, … H. McNeill·bioRxiv·May 26, 2022
- The fluid membrane determines mechanics of red blood cell extracellular vesicles and is softened in hereditary spherocytosis
D. Vorselen, S. van Dommelen, … W. Roos·bioRxiv·Nov 1, 2017
- Non-uniform distribution of myosin-mediated forces governs red blood cell membrane curvature through tension modulation
H. Alimohamadi, A. S. Smith, … P. Rangamani·bioRxiv·Nov 24, 2019
- The membrane skeleton density of red blood cells in MYH9-related disease patients is decreased
Shaopeng Sun, Jiabin Pan, … Xiang‐dong Li·Bioscience ReportsPeer-reviewed·Sep 17, 2026
- Structure, dynamics and assembly of the ankyrin complex on human red blood cell membrane
- Emerin oligomerization and nucleoskeletal coupling at the nuclear envelope regulate nuclear mechanics against stress
A. M. Fernandez, M. Bautista, … F. Pinaud·bioRxiv·Feb 12, 2021