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Base Editing of HBG1 and HBG2 Promoters for Sickle Cell Disease - PubMed

19 hours ago
  • #fetal hemoglobin
  • #base editing
  • #sickle cell disease
  • Ristoglogene autogetemcel (risto-cel) uses base-edited CD34+ hematopoietic stem and progenitor cells to target HBG1/HBG2 promoters and inhibit BCL11A binding, switching hemoglobin production from sickle hemoglobin (HbS) to fetal hemoglobin (HbF).
  • In a phase 1-2 study of 31 sickle cell disease patients aged 12-35 with ≥4 severe vaso-occlusive crises in the previous 2 years, after myeloablative conditioning, a single infusion of risto-cel led to neutrophil engraftment at median 17.5 days and platelet engraftment at 19 days.
  • At 6 months, mean on-target edited alleles were 67.4%, mean HbF exceeded 60% of total hemoglobin, and HbS was below 40%, maintained during follow-up, with no severe vaso-occlusive crises reported >60 days post-transfusion.
  • Adverse events included grade ≥3 events in 87% and serious events in 39% of patients, with one death from idiopathic pneumonia syndrome, but treatment showed durable HbF expression and HbS reduction.
  • Results support further investigation of risto-cel for sickle cell disease treatment, indicating potential efficacy in preventing severe crises and altering hemoglobin profiles.