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Blood, 1 May 2002, Vol. 99, No. 9, pp. 3144-3150
CLINICAL OBSERVATIONS, INTERVENTIONS, AND THERAPEUTIC TRIALS
Moyamoya syndrome in childhood sickle cell disease: a predictive
factor for recurrent cerebrovascular events
Scott R. Dobson,
Kenton R. Holden,
Paul J. Nietert,
Joel K. Cure,
Joseph H. Laver,
Deborah Disco, and
Miguel R. Abboud
From the Departments of Neurology, Pediatrics, and
Radiology and the Center for Health Care Research, Medical University
of South Carolina, Charleston, SC; and the Department of Pediatrics,
Medical College of Virginia, Richmond, VA.
We conducted a retrospective study to determine whether the
presence of moyamoya collaterals influenced the risk of recurrence of
cerebrovascular events (CVEs: stroke or transient ischemic attack) in
patients with sickle cell disease placed on chronic transfusions after
a stroke. Forty-three patients with homozygous sickle cell anemia
(HbSS) and 1 with HbSOArab (16 females, 28 males) who had
suffered strokes while under the age of 18 were studied. All patients
had been on transfusions aimed at maintaining the sickle hemoglobin
(HbS) level below 30%. They were followed for a mean of 6.6 years (2.2 to 20.4 years). The presence of collaterals was diagnosed based on
either magnetic resonance angiography or conventional angiography.
Eighteen (41%) of the 44 patients suffered recurrent CVEs. Nineteen
(43%) (6 females, 13 males) patients had moyamoya collaterals. Eleven
(58%) of these 19 experienced 21 total recurrent CVEs, including 4 strokes in 4 patients (21%). In comparison, 7 (28%) of 25 patients
without moyamoya collaterals experienced 9 recurrent CVEs
(P < .05) with only 1 recurrent stroke (4%). Moyamoya
patients were also more likely to have 2 recurrent CVEs (42% vs 8%,
P < .05) as well as poorer neuropsychological testing
results. A proportional hazards regression analysis indicated that
patients with moyamoya were more than twice as likely to incur a
subsequent CVE (hazard ratio, 2.40; 95% confidence interval, 0.85, 6.75). We conclude that up to 41% of patients with sickle cell disease
experience recurrent CVEs after an initial stroke despite chronic
transfusions and that the risk of recurrence is significantly higher
for those who have moyamoya collaterals.
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