{Reference Type}: Journal Article
{Title}: Development of a mouse embryonic stem cell model for investigating the functions of the linker histone H1-4.
{Author}: Abu Alhaija AA;Lone IN;Sekeroglu EO;Batur T;Angelov D;Dimitrov S;Hamiche A;Firat Karalar EN;Ercan ME;Yagci T;Alotaibi H;Diril MK;
{Journal}: FEBS Open Bio
{Volume}: 14
{Issue}: 2
{Year}: 2024 02 14
{Factor}: 2.792
{DOI}: 10.1002/2211-5463.13750
{Abstract}: The linker histone H1 C-terminal domain (CTD) plays a pivotal role in chromatin condensation. De novo frameshift mutations within the CTD coding region of H1.4 have recently been reported to be associated with Rahman syndrome, a neurological disease that causes intellectual disability and overgrowth. To investigate the mechanisms and pathogenesis of Rahman syndrome, we developed a cellular model using murine embryonic stem cells (mESCs) and CRISPR/Cas9 genome engineering. Our engineered mES cells facilitate detailed investigations, such as H1-4 dynamics, immunoprecipitation, and nuclear localization; in addition, we tagged the mutant H1-4 with a photoactivatable GFP (PA-GFP) and an HA tag to facilitate pulldown assays. We anticipate that these engineered cells could also be used for the development of a mouse model to study the in vivo role of the H1-4 protein.