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PLoS Genet ; 17(4): e1009523, 2021 04.
Article in English | MEDLINE | ID: mdl-33857133

ABSTRACT

The comorbid association of autoimmune diseases with cancers has been a major obstacle to successful anti-cancer treatment. Cancer survival rate decreases significantly in patients with preexisting autoimmunity. However, to date, the molecular and cellular profiles of such comorbidities are poorly understood. We used Aicardi-Goutières syndrome (AGS) as a model autoimmune disease and explored the underlying mechanisms of genome instability in AGS-associated-gene-deficient patient cells. We found that R-loops are highly enriched at transcription-replication conflict regions of the genome in fibroblast of patients bearing SAMHD1 mutation, which is the AGS-associated-gene mutation most frequently reported with tumor and malignancies. In SAMHD1-depleted cells, R-loops accumulated with the concomitant activation of DNA damage responses. Removal of R-loops in SAMHD1 deficiency reduced cellular responses to genome instability. Furthermore, downregulation of SAMHD1 expression is associated with various types of cancer and poor survival rate. Our findings suggest that SAMHD1 functions as a tumor suppressor by resolving R-loops, and thus, SAMHD1 and R-loop may be novel diagnostic markers and targets for patient stratification in anti-cancer therapy.


Subject(s)
Autoimmune Diseases of the Nervous System/genetics , Autoimmune Diseases/genetics , Genomic Instability/genetics , Nervous System Malformations/genetics , SAM Domain and HD Domain-Containing Protein 1/genetics , Autoimmune Diseases/immunology , Autoimmune Diseases/pathology , Autoimmune Diseases of the Nervous System/immunology , Autoimmune Diseases of the Nervous System/pathology , Cell Line, Tumor , DNA Damage/genetics , DNA Replication/genetics , Fibroblasts/metabolism , Genome, Human/genetics , Humans , Mutation/genetics , Neoplasms/genetics , Neoplasms/therapy , Nervous System Malformations/immunology , Nervous System Malformations/pathology , R-Loop Structures/genetics , SAM Domain and HD Domain-Containing Protein 1/ultrastructure , Transcription, Genetic/genetics , Transfection
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