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SIT

Silent Cerebral Infarct Multi-Center Clinical Trial - Controlled Trial of Transfusions for Silent Cerebral Infarcts in Sickle Cell Anemia

Year of Publication: 2014

Authors: Michael R. DeBaun, Mae Gordon, Robert C. McKinstry, ..., James F. Casella

Journal: New England Journal of Medicine

Citation: N Engl J Med 2014;371:699-710


Clinical Question

Does regular blood transfusion therapy reduce the incidence of recurrence of cerebral infarct (stroke or new/enlarged silent cerebral infarct) in children with sickle cell anemia who have silent cerebral infarcts?

Bottom Line

Regular blood transfusion therapy significantly reduced the incidence of infarct recurrence by 58% (from 14% to 6%) in children with sickle cell anemia and silent cerebral infarcts, with a number needed to treat of 13 for 3 years to prevent one recurrence. Transfusion also reduced rates of vaso-occlusive pain, acute chest syndrome, priapism, and avascular necrosis.

Major Points

  • Primary endpoint (infarct recurrence) occurred in 6% of transfusion group vs 14% of observation group
  • Incidence rate ratio 0.41 (95% CI 0.12-0.99, P=0.04), representing 58% relative risk reduction
  • NNT for 3 years to prevent one infarct recurrence: 13
  • Strokes occurred in 1 transfusion patient vs 7 observation patients
  • New or enlarged silent cerebral infarcts: 5 transfusion vs 7 observation
  • TIAs occurred only in observation group (3 patients)
  • Significant reductions in vaso-occlusive pain (IRR 0.41), acute chest syndrome (IRR 0.13), priapism (IRR 0.13), and avascular necrosis (IRR 0.22)
  • No deaths occurred during the trial
  • Alloimmunization rate was 0.278 per 100 units with antigen-matched transfusions
  • Younger age, recurring headaches, and higher reticulocyte count were associated with infarct recurrence

Design

Study Type: Multicenter, randomized, single-blind clinical trial

Randomization: 1

Blinding: Single-blind. Neuroradiology and neurology committees adjudicating MRI findings and neurologic events were unaware of study-group assignments. Participants and treating physicians were not blinded.

Enrollment Period: December 2004 - May 2010

Follow-up Duration: Median 3 years

Centers: 29

Countries: United States, Canada, France, United Kingdom

Sample Size: 196

Analysis: Intention-to-treat. Incidence rate ratio with exact 95% CI using bootstrap methods (10,000 replications). P values estimated using permutation test. Logistic regression to adjust for baseline imbalances and identify risk factors. Sample size calculated for 85% power to detect ≥86% decrease in primary endpoint.


Inclusion Criteria

  • Age 5-15 years
  • Confirmed diagnosis of hemoglobin SS or hemoglobin S-β0 thalassemia
  • At least one infarct-like lesion on screening MRI (≥3 mm in one dimension, visible in two planes on FLAIR T2-weighted images)
  • Adjudicated as silent cerebral infarct (normal neurologic examination or abnormality not explained by lesion location)
  • Written informed consent from parents/guardians and assent from participants

Exclusion Criteria

  • History of focal neurologic deficit associated with an infarct on brain MRI (overt stroke)
  • Seizure disorder
  • Treatment with hydroxyurea in previous 3 months
  • History of regular transfusion therapy
  • Imaging or nonimaging transcranial Doppler measurement above study-defined thresholds

Arms

FieldControlTransfusion
InterventionStandard care with no specific treatment for silent cerebral infarcts, including no hydroxyurea therapy. Evaluated quarterly. No transfusions specifically for silent cerebral infarcts (though 32% received episodic transfusions and 6 crossed over to regular transfusions). 14% started hydroxyurea during trial due to disease severity.Regular blood transfusion therapy approximately monthly. Target hemoglobin concentration >9.0 g/dL. Target hemoglobin S concentration ≤30% of total hemoglobin. Ferritin monitored before each transfusion; chelation initiated if ferritin >1500 ng/mL for ≥2 consecutive months. 90 of 99 started transfusions within 4 weeks of assignment. 95% of 3236 transfusions had intervals ≤38 days. Median hemoglobin S level was 30.1%.
DurationMedian 3 years (289 patient-years total)Median 3 years (304 patient-years total)

Outcomes

OutcomeTypeControlInterventionHR / OR / RRP-value
Recurrence of infarct or hemorrhage as determined by neuroimaging, clinical evidence of permanent neurologic injury, or both. Includes stroke, new silent cerebral infarct (≥3 mm, visible in two planes), or enlarged silent cerebral infarct (increased by ≥3 mm in any dimension).Primary14% (14/97); 4.8 events per 100 person-years at risk6% (6/99); 2.0 events per 100 person-years at risk8.37%0.04
StrokeSecondary7/97 (7%)1/99 (1%)
New or enlarged silent cerebral infarctSecondary7/97 (7%)5/99 (5%)
Transient ischemic attack (TIA)Secondary3/97 (3%); 2 TIA only, 1 TIA then stroke0/99 (0%)
All neurologic events (infarct recurrence + TIA)Secondary5.6 per 100 person-years2.0 per 100 person-years0.02
Full-scale IQ change (baseline to exit)SecondaryNo significant changeNo significant changeNS within or between groups
BRIEF (Behavior Rating Inventory of Executive Function) changeSecondaryNo significant changeNo significant changeNS within or between groups
DeathAdverse00
Vaso-occlusive painAdverse56 patients with ≥1 event; 102.21 per 100 person-years32 patients with ≥1 event; 41.58 per 100 person-years0.004
Acute chest syndromeAdverse24 patients; 14.35 per 100 person-years5 patients; 1.81 per 100 person-years<0.001
Priapism (males only)Adverse7/52 males; 6.65 per 100 person-years1/59 males; 0.84 per 100 person-years0.02
Symptomatic avascular necrosis of hipAdverse6 patients; 2.25 per 100 person-years1 patient; 0.49 per 100 person-years0.02
HeadacheAdverse30 patients; 32.34 per 100 person-years24 patients; 25.15 per 100 person-years0.40
Blood transfusion reactionAdverse1/31 transfused patients; 1.66 per 100 person-years15/90 patients (17%); 8.85 per 100 person-years (25 total reactions: 13 allergic, 8 febrile nonhemolytic)0.05
Ferritin >1500 ng/mLAdverse3/31 transfused; 37.07 per 100 person-years76/90 patients; 534.70 per 100 person-years<0.001
AlloimmunizationAdverse04 patients with 9 alloantibodies (anti-C ×2, anti-V ×2, anti-FyA, anti-e, anti-S, anti-JK-b, anti-Wra); rate 0.278 per 100 units
Central venous catheter complicationsAdverse11 catheters placed; 1 infection, 2 requiring replacement

Subgroup Analysis

Risk factors for infarct recurrence in logistic regression: Transfusion group vs observation (OR 0.31, 95% CI 0.10-0.93, P=0.04). Younger age (OR 1.41 per year younger, 95% CI 1.12-1.78, P=0.004). History of recurring headaches (OR 4.33, 95% CI 1.50-13.06, P=0.007). Higher steady-state reticulocyte count (OR 1.11 per 1% increase, 95% CI 1.01-1.22, P=0.04).


Criticisms

  • 15% crossover rate from transfusion to observation (9 declined immediately, 6 crossed over later)
  • 32% of observation group received episodic transfusions; 6 crossed over to regular transfusions
  • Open-label to participants and treating physicians
  • Not powered to detect changes in IQ or cognitive outcomes
  • Duration of blood transfusion therapy for secondary prevention unknown; minimum 3 years suggested
  • Excluded children on hydroxyurea, with elevated TCD, or receiving transfusions for primary stroke prevention
  • Results not directly applicable to all children with sickle cell anemia due to exclusion criteria
  • Iron overload requiring chelation is a significant burden of transfusion therapy
  • Two participants with TCD above eligibility threshold were inadvertently randomized due to programming error

Funding

National Institute of Neurological Disorders and Stroke (5U01NS042804, 3U01NS042804 American Recovery Reinvestment Act supplementary grant); Institute of Clinical and Translational Sciences, National Center for Research Resources, National Center for Advancing Translational Sciences, Clinical and Translational Research; NIH Roadmap for Medical Research (UL1TR000448 to Washington University, UL1TR001079 to Johns Hopkins University, UL1TR000003 to Children's Hospital of Philadelphia); Research and Development in the National Health Service, United Kingdom.

Based on: SIT (New England Journal of Medicine, 2014)

Authors: Michael R. DeBaun, Mae Gordon, Robert C. McKinstry, ..., James F. Casella

Citation: N Engl J Med 2014;371:699-710

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