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The Clinical Effectiveness and Cost-Effectiveness of Primary Stroke Prevention in Children with Sickle Cell Disease: A Systematic Review and Economic Evaluation

The Clinical Effectiveness and Cost-Effectiveness of Primary Stroke Prevention in Children with Sickle Cell Disease: A Systematic Review and Economic Evaluation

Health Technology Assessment - NIHR Evaluation, Trials and Studies Coordinating Centre (UK)

Version: November 2012

Background

Sickle cell disease is a recessive genetic blood disorder, caused by a mutation in the beta-globin gene. This mutation results in an altered haemoglobin molecule that polymerises when deoxygenated and damages red cells, which adopt the characteristic sickle shape. Their abnormal shape and decreased flexibility means that they are more likely to obstruct small blood vessels, reducing the amount of oxygen delivered to the lungs, brain and other tissues, and causing vascular endothelial damage.

Assessment of clinical effectiveness

A total of 1337 non-duplicate records were identified by the search strategy (see Appendix 1) and subsequently screened for inclusion in the review. No trials were identified that evaluated the efficacy of hydroxycarbamide or BMT as primary stroke prevention strategies. Two RCTs, made comparisons between blood transfusion and standard care and were included in the review. Data from these trials were published in peer-reviewed journals. A number of papers relating to these two RCTs were also identified.

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