{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,29]],"date-time":"2026-05-29T14:45:03Z","timestamp":1780065903485,"version":"3.54.0"},"reference-count":62,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2023,1,10]],"date-time":"2023-01-10T00:00:00Z","timestamp":1673308800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Immunol."],"abstract":"<jats:sec><jats:title>Introduction<\/jats:title><jats:p>The granulocyte colony-stimulating factor receptor (G-CSFR), encoded by the <jats:italic>CSF3R<\/jats:italic> gene, is involved in the production and function of neutrophilic granulocytes. Somatic mutations in <jats:italic>CSF3R<\/jats:italic> leading to truncated G-CSFR forms are observed in acute myeloid leukemia (AML), particularly those subsequent to severe chronic neutropenia (SCN), as well as in a subset of patients with other leukemias.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>This investigation introduced equivalent mutations into the zebrafish <jats:italic>csf3r<\/jats:italic> gene <jats:italic>via<\/jats:italic> genome editing and used a range of molecular and cellular techniques to understand the impact of these mutations on immune cells across the lifespan.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Zebrafish harboring truncated G-CSFRs showed significantly enhanced neutrophil production throughout successive waves of embryonic hematopoiesis and a neutrophil maturation defect in adults, with the mutations acting in a partially dominant manner.<\/jats:p><\/jats:sec><jats:sec><jats:title>Discussion<\/jats:title><jats:p>This study has elucidated new insights into the impact of G-CSFR truncations throughout the life-course and created a <jats:italic>bone fide<\/jats:italic> zebrafish model for further investigation.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fimmu.2022.1095453","type":"journal-article","created":{"date-parts":[[2023,1,10]],"date-time":"2023-01-10T20:39:44Z","timestamp":1673383184000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":2,"title":["Leukemia-associated truncation of granulocyte colony-stimulating factor receptor impacts granulopoiesis throughout the 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