ArticleHaematologica2016
Long-term serial xenotransplantation of juvenile myelomonocytic leukemia recapitulates human disease in Rag2-/-γc-/- mice.
Article in Haematologica, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers.
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Who cites it
15 citing papers in PubMed, 26 citations in OpenAlex.
- Novel humanized loss-of-function NF1 mouse model of juvenile myelomonocytic leukemia.Blood advances · 2026Article
- Oncogenic and microenvironmental signals drive cell type specific apoptosis resistance in juvenile myelomonocytic leukemia.Cell death & disease · 2025Article
- Progression and perspectives in disease modeling for Juvenile myelomonocytic leukemia.Medical oncology (Northwood, London, England) · 2024Review
- Progression and perspectives in disease modeling for Juvenile myelomonocytic leukemia.Medical oncology (Northwood, London, England) · 2024Review
- BH3 mimetics and azacitidine show synergistic effects on juvenile myelomonocytic leukemia.Leukemia · 2024Article
- Epigenetic Profiling ofCancers · 2023Article
- Review
- Long non-coding RNAs as novel therapeutic targets in juvenile myelomonocytic leukemia.Scientific reports · 2021Observational
- BCL-XL expression is essential for human erythropoiesis and engraftment of hematopoietic stem cells.Cell death & disease · 2020Article
- Proteomic Analysis of an Induced Pluripotent Stem Cell Model Reveals Strategies to Treat Juvenile Myelomonocytic Leukemia.Journal of proteome research · 2020Article
- Single-Center Experience With Epigenetic Treatment for Juvenile Myelomonocytic Leukemia.Frontiers in oncology · 2020Article
- Azacitidine is effective for targeting leukemia-initiating cells in juvenile myelomonocytic leukemia.Leukemia · 2019Article
- Article
- Mice expressing KrasG12D in hematopoietic multipotent progenitor cells develop neonatal myeloid leukemia.The Journal of clinical investigation · 2017Article
- Article
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Authors and funding
9 authors at 3 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Juvenile myelomonocytic leukemia is a clonal malignant disease affecting young children. Current cure rates, even with allogeneic hematopoietic stem cell transplantation, are no better than 50%-60%. Pre-clinical research on juvenile myelomonocytic leukemia is urgently needed for the identification of novel therapies but is hampered by the unavailability of culture systems. Here we report a xenotransplantation model that allows long-term in vivo propagation of primary juvenile myelomonocytic leukemia cells. Persistent engraftment of leukemic cells was achieved by intrahepatic injection of 1×10(6) cells into newborn Rag2(-/-)γc(-/-) mice or intravenous injection of 5×10(6) cells into 5-week old mice. Key characteristics of juvenile myelomonocytic leukemia were reproduced, including cachexia and clonal expansion of myelomonocytic progenitor cells that infiltrated bone marrow, spleen, liver and, notably, lung. Xenografted leukemia cells led to reduced survival of recipient mice. The stem cell character of juvenile myelomonocytic leukemia was confirmed by successful serial transplantation that resulted in leukemia cell propagation for more than one year. Independence of exogenous cytokines, low donor cell number and slowly progressing leukemia are advantages of the model, which will serve as an important tool to research the pathophysiology of juvenile myelomonocytic leukemia and test novel pharmaceutical strategies such as DNA methyltransferase inhibition.
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