The mouse Klf1 Nan variant impairs nuclear condensation and erythroid maturation
Ileana Cantú, Harmen J.G. van de WerkeniD, Nynke Gillemans, Ralph StadhoudersiD, Steven Heshusius, Alex MaasiD, Fatemehsadat EsteghamatiD, Zeliha ÖzgüriD, Wilfred F. J. van IJckeniD, Frank G. GrosveldiD, Marieke v. LinderniD, Sjaak PhilipseniD, Thamar B. van Dijk
Abstract
Kruppel-like factor 1 (KLF1) is an essential transcription factor for erythroid development, as demonstrated by Klf1 knockout mice which die around E14 due to severe anemia. In humans, >65 KLF1 variants, causing different erythroid phenotypes, have been described. The Klf1 Nan variant, a single amino acid substitution (p.E339D) in the DNA binding domain, causes hemolytic anemia and is dominant over wildtype KLF1. Here we describe the effects of the Nan variant during fetal development. We show that Nan embryos have defects in erythroid maturation. RNA-sequencing of the Nan fetal liver cells revealed that Exportin 7 (Xpo7) was among the ~780 deregulated genes. This nuclear exportin is implicated in terminal erythroid differentiation; in particular it is involved in nuclear condensation. Indeed, KLF1 Nan fetal liver cells had larger nuclei and reduced chromatin condensation. Knockdown of XPO7 in wildtype erythroid cells caused a similar phenotype. We conclude that reduced expression of XPO7 is partially responsible for the erythroid defects observed in Nan erythroid cells.
02 /Specification & Classification
Click any tag to browse matching papers- Citations
- 15
- FWCI
- 0.83
- Field percentile
- top 32%
- Organism
- Assay
- Research Focus
- Approach
Reproducibility ledger
Nobody has reported trying to reproduce this paper yet. Did you replicate an experiment or run the code? Your report saves the next lab a week.
Sign in to file a reproducibility report.
Related papers
- A Krüppel-like factor 1 (KLF1) mutation associated with severe congenital dyserythropoietic anemia alters its DNA-binding specificity
Klaudia Kulczyńska-Figurny, James J. Bieker, Mirosława Siatecka·Molecular and Cellular BiologyPeer-reviewed·Dec 9, 2019
- STING and VDAC inhibitors attenuate inflammation and ineffective erythropoiesis caused by an altered metabolome in the Nan (EKLF/E339D) mouse model of neonatal anemia
Tasleem Arif, Kaustav Mukherjee, … James J. Bieker·bioRxiv·Dec 12, 2025
- A Positive Regulatory Feedback Loop Between 1 EKLF/ KLF1 and TAL1/SCL2 Sustaining the Erythropoiesis
C.-H. Hung, Y.-S. Huang, … C.-K. J. Shen·bioRxiv·Apr 8, 2021
- Modelling the Erythroblastic Island Niche of Dyserythropoietic Anaemia Type IV patients using Induced Pluripotent Stem Cells
A. May, T. Ventura, … L. M. Forrester·bioRxiv·Feb 2, 2023
- The Role of DOT1L Methyltransferase Activity in Fetal Hematopoiesis
Carrie A. Malcom, Joanna Piasecka‐Srader, … Patrick E. Fields·bioRxiv·Sep 30, 2020
- Requirement for Anti-Apoptotic MCL-1 during Early Erythropoiesis
Meghan E. Turnis, Ewa K. Kaminska, … Joseph T. Opferman·BloodPeer-reviewed·Apr 8, 2021