ArticleLeukemia2017
Establishment of a congenital amegakaryocytic thrombocytopenia model and a thrombocyte-specific reporter line in zebrafish.
Article in Leukemia, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 17 papers.
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Who cites it
17 citing papers in PubMed, 31 citations in OpenAlex.
- The endothelial scavenger receptorbioRxiv : the preprint server for biology · 2026Article
- Structural insights into antibody responses against influenza A virus in its natural reservoir.bioRxiv : the preprint server for biology · 2026Article
- Minoxidil ameliorates myelodysplastic syndrome by targeting Wnt4 while sparing normal hematopoiesis.Cell communication and signaling : CCS · 2025Article
- Decitabine facilitates thrombopoiesis independent of the thrombopoietin receptor in zebrafish.Blood science (Baltimore, Md.) · 2025Article
- Learning from Zebrafish Hematopoiesis.Advances in experimental medicine and biology · 2023Review
- Splicing factor deficits render hematopoietic stem and progenitor cells sensitive to STAT3 inhibition.Cell reports · 2022Article
- Establishment of a Bernard-Soulier syndrome model in zebrafish.Haematologica · 2022Article
- Genetics of inherited thrombocytopenias.Blood · 2022Review
- Erythropoietin receptor contributes to thrombopoietin receptor (Mpl)-independent thrombocytopoiesis in zebrafish.Leukemia · 2022Article
- Zebrafish models of inflammation in hematopoietic development and disease.Frontiers in cell and developmental biology · 2022Review
- Slc20a1b is essential for hematopoietic stem/progenitor cell expansion in zebrafish.Science China. Life sciences · 2021Article
- Zebrafish Cancer Predisposition Models.Frontiers in cell and developmental biology · 2021Review
- Potential Applications of Fluorescence-Activated Cell Sorting (FACS) and Droplet-Based Microfluidics in Promoting the Discovery of Specific Antibodies for Characterizations of Fish Immune Cells.Frontiers in immunology · 2021Review
- Zebrafish for thrombocytopoiesis- and hemostasis-related researches and disorders.Blood science (Baltimore, Md.) · 2020Review
- Modeling hematopoietic disorders in zebrafish.Disease models & mechanisms · 2019Review
- Establishment of a zebrafish hematological disease model induced by 1,4-benzoquinone.Disease models & mechanisms · 2019Article
- Peering through zebrafish to understand inherited bone marrow failure syndromes.Haematologica · 2019Review
Corrections and comments
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Authors and funding
10 authors at 2 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Mutations in the human myeloproliferative leukemia (MPL) protein gene are known to cause congenital amegakaryocytic thrombocytopenia (CAMT). The prognosis of this heritable disorder is poor and bone marrow transplantation is the only effective treatment. Here, by using the TALEN (transcription activator-like effector nuclease) technology, we created a zebrafish mpl mutant to model human CAMT. Disruption of zebrafish mpl lead to a severe reduction in thrombocytes and a high bleeding tendency, as well as deficiencies in adult hematopoietic stem/progenitor cells. We further demonstrated that thrombocytopenia in mpl mutant zebrafish was caused by impaired Tpo/Mpl/Jak2 signaling, resulting in reduced proliferation of thrombocyte precursors. These results indicate that mpl mutant zebrafish develop thrombocytopenia resembling the human CAMT. To utilize fully zebrafish to study thrombocyte biology and thrombocytopenia disorders, we generated a transgenic reporter line Tg(mpl:eGFP)smu4, in which green fluorescent protein (GFP) expression was driven by the mpl promoter. Detailed characterization of Tg(mpl:eGFP)smu4 fish confirmed that the thrombocyte lineage was specifically marked by GFP expression. In conclusion, we generated the first transmissible congenital thrombocytopenia zebrafish model mimicking human CAMT and a thrombocyte-specific transgenic line. Together with Tg(mpl:eGFP)smu4, mpl mutant zebrafish provide a useful tool for drug screening and study of thrombocytopoiesis.
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