Association of circulating transcriptomic profiles with mortality in sickle cell disease

Blood. 2017 Jun 1;129(22):3009-3016. doi: 10.1182/blood-2016-11-752279. Epub 2017 Apr 3.

Abstract

Sickle cell disease (SCD) complications are associated with increased morbidity and risk of mortality. We sought to identify a circulating transcriptomic profile predictive of these poor outcomes in SCD. Training and testing cohorts consisting of adult patients with SCD were recruited and prospectively followed. A pathway-based signature derived from grouping peripheral blood mononuclear cell transcriptomes distinguished 2 patient clusters with differences in survival in the training cohort. These findings were validated in a testing cohort in which the association between cluster 1 molecular profiling and mortality remained significant in a fully adjusted model. In a third cohort of West African children with SCD, cluster 1 differentiated SCD severity using a published scoring index. Finally, a risk score composed of assigning weights to cluster 1 profiling, along with established clinical risk factors using tricuspid regurgitation velocity, white blood cell count, history of acute chest syndrome, and hemoglobin levels, demonstrated a higher hazard ratio for mortality in both the training and testing cohorts compared with clinical risk factors or cluster 1 data alone. Circulating transcriptomic profiles are a powerful method to risk-stratify severity of disease and poor outcomes in both children and adults, respectively, with SCD and highlight potential associated molecular pathways.

Publication types

  • Validation Study

MeSH terms

  • Acute Chest Syndrome / genetics
  • Adult
  • Anemia, Sickle Cell / blood
  • Anemia, Sickle Cell / genetics*
  • Anemia, Sickle Cell / mortality
  • Child
  • Cohort Studies
  • Female
  • Hemoglobins / metabolism
  • Humans
  • Kaplan-Meier Estimate
  • Leukocyte Count
  • Male
  • Middle Aged
  • Prognosis
  • Prospective Studies
  • Risk Factors
  • Transcriptome
  • Tricuspid Valve Insufficiency / genetics
  • Young Adult

Substances

  • Hemoglobins