Genetic Modifiers of Stroke in Patients with Sickle Cell Disease-A Scoping Review

Int J Mol Sci. 2024 Jun 7;25(12):6317. doi: 10.3390/ijms25126317.

Abstract

Sickle cell disease (SCD) clinically manifests itself with a myriad of complications. Stroke, both ischemic and hemorrhagic, as well as silent white matter changes, occurs at a relatively high prevalence. Understanding why and in whom stroke is most likely to occur is critical to the effective prevention and treatment of individuals with SCD. Genetic studies, including genome- and exome-wide association studies (GWAS and EWAS), have found several key modifiers associated with increased stroke/stroke risk in SCD via mechanisms including Hemoglobin F (HbF) modulation, inflammation, cellular adhesion, endothelial disruption, and hemolysis. We present a review on the modifiers that have most clearly demonstrated an association to date. More studies are needed to validate other potential polymorphisms and identify new ones. Incorporating gene-focused screenings in clinical care could provide avenues for more targeted, more effective, and less toxic prevention of stroke in this population. The data from this review will be used to inform the initial GWAS performed by the International Hemoglobinopathy Research Network (INHERENT) consortium.

Keywords: cerebral vasculopathy; genetic modifiers; sickle cell disease; stroke.

Publication types

  • Review

MeSH terms

  • Anemia, Sickle Cell* / complications
  • Anemia, Sickle Cell* / genetics
  • Fetal Hemoglobin / genetics
  • Fetal Hemoglobin / metabolism
  • Genes, Modifier
  • Genetic Predisposition to Disease*
  • Genome-Wide Association Study*
  • Humans
  • Stroke* / genetics

Substances

  • Fetal Hemoglobin

Grants and funding

This work was partially supported by H&TRC, FCT/MCTES grant number (UIDB/05608/2020 and UIDP/05608/2020) and the Dana-Farber/Boston Children’s Cancer and Blood Disorders Center Talent Development Award.