Intracranial Aneurysms in Sickle-Cell Disease Are Associated With the Hemoglobin SS Genotype But Not With Moyamoya Syndrome

Stroke. 2016 Jul;47(7):1710-3. doi: 10.1161/STROKEAHA.116.012664. Epub 2016 Jun 14.

Abstract

Background and purpose: Intracranial aneurysms and aneurysmal subarachnoid hemorrhage may occur more frequently in sickle-cell disease (SCD), and this could be related to the sickle genotype and moyamoya syndrome seen in SCD.

Methods: Records from a total of 1002 patients with SCD attending 2 specialized adult hematologic services were retrospectively reviewed. We analyzed data of a cohort of 767 patients attending 1 SCD clinic between 2002 and 2013 and of 235 patients from the other clinic who have had neurovascular imaging between 2007 and 2014.

Results: We identified 4 patients in the cohort who had an aneurysmal subarachnoid hemorrhage during 9063 patient-years. The highest incidence rate was seen among women in the age group 30 to 39 years with the hemoglobin SS (HbSS) genotype (440 per 100 000 patient-years). Unruptured intracranial aneurysms were found in 20 of the 324 patients, who had imaging data; the prevalence was significantly higher in patients with HbSS genotype compared with other sickle genotypes with the highest prevalence (15%) observed in women in the age group 30 to 39 years. Fifty-one HbSS patients had a moyamoya vasculopathy, but only 3 of these had concomitant intracranial aneurysms.

Conclusions: Intracranial aneurysms are common in HbSS SCD. There was also a trend toward more common occurrence of aneurysmal subarachnoid hemorrhage in HbSS; women in the age group 30 to 39 years were most at risk. There was no correlation between the occurrence of intracranial aneurysms and moyamoya syndrome.

Keywords: anemia, sickle cell; intracranial aneurysm; moyamoya disease; stroke; subarachnoid hemorrhage.

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Aged, 80 and over
  • Anemia, Sickle Cell / diagnostic imaging
  • Anemia, Sickle Cell / epidemiology*
  • Anemia, Sickle Cell / genetics
  • Brain / diagnostic imaging
  • Cerebral Angiography
  • Child
  • Child, Preschool
  • Comorbidity
  • Female
  • Genetic Predisposition to Disease
  • Genotype*
  • Hemoglobin, Sickle / genetics*
  • Humans
  • Incidence
  • Infant
  • Infant, Newborn
  • Intracranial Aneurysm / diagnostic imaging
  • Intracranial Aneurysm / epidemiology*
  • Intracranial Aneurysm / genetics
  • Magnetic Resonance Angiography
  • Male
  • Middle Aged
  • Moyamoya Disease / diagnostic imaging
  • Moyamoya Disease / epidemiology*
  • Moyamoya Disease / genetics
  • Prevalence
  • Retrospective Studies
  • Young Adult

Substances

  • Hemoglobin, Sickle