Tracy A. Willson

Differential in vivo roles of Mpl cytoplasmic tyrosine residues in murine hematopoiesis and myeloproliferative disease

Kira Behrens, Maria Kauppi, Elizabeth M. Viney, Andrew J. Kueh et autres

Thrombopoietin (Tpo), which binds to its specific receptor, the Mpl protein, is the major cytokine regulator of megakaryopoiesis and circulating platelet number. Tpo binding to Mpl triggers activation of Janus kinase 2 (Jak2) and phosphorylation of the receptor, as well as activation …

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BAF complex-mediated chromatin relaxation is required for establishment of X chromosome inactivation

Andrew Keniry, Natasha Jansz, Linden J. Gearing, Iromi Wanigasuriya et autres

The process of epigenetic silencing, while fundamentally important, is not yet completely understood. Here we report a replenishable female mouse embryonic stem cell (mESC) system, Xmas, that allows rapid assessment of X chromosome inactivation (XCI), the epigenetic silencing mechanism of one of …

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3104 – CLONAL MULTI-OMICS METHODS SIS-SEQ AND SIS-SKEW REVEAL BCOR AS A NEGATIVE REGULATOR OF EMERGENCY DENDRITIC CELL DEVELOPMENT

Shalin H. Naik, Luyi Tian, Sara Tomei, Jaring Schreuder et autres

A functional genetic screen identifies aurora kinase b as an essential regulator of Sox9-positive mouse embryonic lung progenitor cells

Casey Ah-Cann, Verena C. Wimmer, Clare E. Weeden, Claire Marceaux et autres

Development of a branching tree in the embryonic lung is crucial for the formation of a fully mature functional lung at birth. Sox9+ cells present at the tip of the primary embryonic lung endoderm are multipotent cells responsible for branch formation and …

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SMCHD1's ubiquitin-like domain is required for N-terminal dimerization and chromatin localization

Alexandra D. Gurzau, Christopher R. Horne, Yee-Foong Mok, Megan Iminitoff et autres

Structural maintenance of chromosomes flexible hinge domain-containing 1 (SMCHD1) is an epigenetic regulator that mediates gene expression silencing at targeted sites across the genome. Our current understanding of SMCHD1's molecular mechanism, and how substitutions within SMCHD1 lead to the diseases, facioscapulohumeral muscular …

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Clonal multi-omics reveals Bcor as a negative regulator of emergency dendritic cell development

Luyi Tian, Sara Tomei, Jaring Schreuder, Tom Weber et autres

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A missense mutation in the MLKL brace region promotes lethal neonatal inflammation and hematopoietic dysfunction

Joanne M. Hildebrand, Maria Kauppi, Ian J. Majewski, Zikou Liu et autres

Abstract MLKL is the essential effector of necroptosis, a form of programmed lytic cell death. We have isolated a mouse strain with a single missense mutation,MlklD139V, that alters the two-helix ‘brace’ that connects the killer four-helix bundle and regulatory pseudokinase domains. This …

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Crystal structure of the hinge domain of Smchd1 reveals its dimerization mode and nucleic acid–binding residues

Kelan Chen, Richard W. Birkinshaw, Alexandra D. Gurzau, Iromi Wanigasuriya et autres

Structural maintenance of chromosomes flexible hinge domain containing 1 (SMCHD1) is an epigenetic regulator in which polymorphisms cause the human developmental disorder, Bosma arhinia micropthalmia syndrome, and the degenerative disease, facioscapulohumeral muscular dystrophy. SMCHD1 is considered a noncanonical SMC family member because …

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An Erg-driven transcriptional program controls B cell lymphopoiesis

Ashley P. Ng, Hannah D. Coughlan, Soroor Hediyeh-zadeh, Kira Behrens et autres

B lymphoid development is initiated by the differentiation of hematopoietic stem cells into lineage committed progenitors, ultimately generating mature B cells. This highly regulated process generates clonal immunological diversity via recombination of immunoglobulin V, D and J gene segments. While several transcription …

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HBO1 is required for the maintenance of leukaemia stem cells

Laura MacPherson, Juliana Anokye, Miriam M. Yeung, Enid Y.N. Lam et autres

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An Erg driven transcriptional program controls B-lymphopoiesis

Ashley P. Ng, Hannah D. Coughlan, Soroor Hediyeh-zadeh, Kira Behrens et autres

Summary/Abstract B-cell development is initiated by the stepwise differentiation of hematopoietic stem cells into lineage committed progenitors, ultimately generating the mature B-cells that mediate protective immunity. This highly regulated process also generates clonal immunological diversity via recombination of immunoglobulin genes. While several …

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Xmas ESC: A new female embryonic stem cell system that reveals the BAF complex as a key regulator of the establishment of X chromosome inactivation

Andrew Keniry, Natasha Jansz, Linden J. Gearing, Iromi Wanigasuriya et autres

Summary Although female pluripotency significantly differs to male, complications with in vitro culture of female embryonic stem cells (ESC) have severely limited the use and study of these cells. We report a replenishable female ESC system, Xmas, that has enabled us to …

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