Impaired Replication Stress Response in Cells from Immunodeficiency Patients Carrying Cernunnos/XLF Mutations
2009

Impaired DNA Repair in Immunodeficiency Patients with Cernunnos/XLF Mutations

publication Evidence: high

Author Information

Author(s): Schwartz Michal, Oren Yifat S., Bester Assaf C., Rahat Ayelet, Sfez Ruthy, Yitzchaik Shlomo, de Villartay Jean-Pierre, Kerem Batsheva

Primary Institution: The Hebrew University, Jerusalem, Israel

Hypothesis

Do Cernunnos/XLF mutations disrupt the ability of patient cells to respond to replication stress conditions?

Conclusion

Cernunnos/XLF mutations in immunodeficiency patients lead to impaired cellular response to replication stress and increased chromosomal instability.

Supporting Evidence

  • Cernunnos/XLF mutated cells show increased sensitivity to replication stress.
  • Impaired DSB repair was observed in Cernunnos/XLF mutated cells under replication stress.
  • Cells with Cernunnos/XLF mutations displayed greater chromosomal instability at fragile sites.

Takeaway

Some people have a gene that helps fix DNA when it gets damaged. If this gene is broken, their cells can't fix the damage properly, which can make them sick.

Methodology

The study analyzed the cellular response to replication stress in patient cells with Cernunnos/XLF mutations and compared them to complemented cells.

Limitations

The study primarily focuses on specific mutations and may not represent all cases of Cernunnos/XLF deficiency.

Participant Demographics

Patients with immunodeficiency and microcephaly due to Cernunnos/XLF mutations.

Statistical Information

P-Value

p<0.05

Statistical Significance

p<0.05

Digital Object Identifier (DOI)

10.1371/journal.pone.0004516

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