Cargando…
Gata1s mutant mice display persistent defects in the erythroid lineage
GATA1 mutations that result in loss of the N-terminal 83 amino acids are a feature of myeloid leukemia in children with Down syndrome, rare familial cases of dyserythropoietic anemia, and a subset of cases of Diamond-Blackfan anemia. The Gata1s mouse model, which expresses only the short GATA1 isofo...
Autores principales: | , , , , |
---|---|
Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
The American Society of Hematology
2022
|
Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10336263/ https://www.ncbi.nlm.nih.gov/pubmed/36350717 http://dx.doi.org/10.1182/bloodadvances.2022008124 |
_version_ | 1785071171927015424 |
---|---|
author | Ling, Te Zhang, Kevin Yang, Jiayue Gurbuxani, Sandeep Crispino, John D. |
author_facet | Ling, Te Zhang, Kevin Yang, Jiayue Gurbuxani, Sandeep Crispino, John D. |
author_sort | Ling, Te |
collection | PubMed |
description | GATA1 mutations that result in loss of the N-terminal 83 amino acids are a feature of myeloid leukemia in children with Down syndrome, rare familial cases of dyserythropoietic anemia, and a subset of cases of Diamond-Blackfan anemia. The Gata1s mouse model, which expresses only the short GATA1 isoform that begins at methionine 84, has been shown to have a defect in hematopoiesis, especially impaired erythropoiesis with expanded megakaryopoiesis, during gestation. However, these mice reportedly did not show any postnatal phenotype. Here, we demonstrate that Gata1s mutant mice display macrocytic anemia and features of aberrant megakaryopoiesis throughout life, culminating in profound splenomegaly and bone marrow fibrosis. These data support the use of this animal model for studies of GATA1 deficiencies. |
format | Online Article Text |
id | pubmed-10336263 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | The American Society of Hematology |
record_format | MEDLINE/PubMed |
spelling | pubmed-103362632023-07-13 Gata1s mutant mice display persistent defects in the erythroid lineage Ling, Te Zhang, Kevin Yang, Jiayue Gurbuxani, Sandeep Crispino, John D. Blood Adv Red Cells, Iron, and Erythropoiesis GATA1 mutations that result in loss of the N-terminal 83 amino acids are a feature of myeloid leukemia in children with Down syndrome, rare familial cases of dyserythropoietic anemia, and a subset of cases of Diamond-Blackfan anemia. The Gata1s mouse model, which expresses only the short GATA1 isoform that begins at methionine 84, has been shown to have a defect in hematopoiesis, especially impaired erythropoiesis with expanded megakaryopoiesis, during gestation. However, these mice reportedly did not show any postnatal phenotype. Here, we demonstrate that Gata1s mutant mice display macrocytic anemia and features of aberrant megakaryopoiesis throughout life, culminating in profound splenomegaly and bone marrow fibrosis. These data support the use of this animal model for studies of GATA1 deficiencies. The American Society of Hematology 2022-11-11 /pmc/articles/PMC10336263/ /pubmed/36350717 http://dx.doi.org/10.1182/bloodadvances.2022008124 Text en © 2023 by The American Society of Hematology. Licensed under Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International (CC BY-NC-ND 4.0), permitting only noncommercial, nonderivative use with attribution. All other rights reserved. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). |
spellingShingle | Red Cells, Iron, and Erythropoiesis Ling, Te Zhang, Kevin Yang, Jiayue Gurbuxani, Sandeep Crispino, John D. Gata1s mutant mice display persistent defects in the erythroid lineage |
title | Gata1s mutant mice display persistent defects in the erythroid lineage |
title_full | Gata1s mutant mice display persistent defects in the erythroid lineage |
title_fullStr | Gata1s mutant mice display persistent defects in the erythroid lineage |
title_full_unstemmed | Gata1s mutant mice display persistent defects in the erythroid lineage |
title_short | Gata1s mutant mice display persistent defects in the erythroid lineage |
title_sort | gata1s mutant mice display persistent defects in the erythroid lineage |
topic | Red Cells, Iron, and Erythropoiesis |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10336263/ https://www.ncbi.nlm.nih.gov/pubmed/36350717 http://dx.doi.org/10.1182/bloodadvances.2022008124 |
work_keys_str_mv | AT lingte gata1smutantmicedisplaypersistentdefectsintheerythroidlineage AT zhangkevin gata1smutantmicedisplaypersistentdefectsintheerythroidlineage AT yangjiayue gata1smutantmicedisplaypersistentdefectsintheerythroidlineage AT gurbuxanisandeep gata1smutantmicedisplaypersistentdefectsintheerythroidlineage AT crispinojohnd gata1smutantmicedisplaypersistentdefectsintheerythroidlineage |