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J Pediatr ; 162(2): 375-80.e1, 2013 Feb.
Artículo en Inglés | MEDLINE | ID: mdl-22974573

RESUMEN

OBJECTIVE: To investigate whether intravenous enzyme replacement therapy (ERT) benefits cognitive function in patients with mucopolysaccharidosis type IH (Hurler syndrome) undergoing hematopoietic cell transplantation (HCT). STUDY DESIGN: Data were obtained for 9 children treated with HCT + ERT (ERT group) and 10 children treated with HCT only (no-ERT group) from neuropsychologic evaluations before HCT and at 1-year and 2-year post-HCT follow-up. RESULTS: At 2 years after HCT, children in the ERT group lost 9.19 fewer IQ points per year compared with children in the no-ERT group (P = .031). Furthermore, the ERT group improved in nonverbal problem solving and processing, whereas the no-ERT group declined, resulting in a difference of 9.44 points per year between the 2 groups (P < .001). CONCLUSION: ERT in association with HCT enhances cognitive outcomes, providing new evidence that ERT is a valuable addition to the standard transplantation protocol. Although the mechanism responsible for this improved outcome is unknown, both direct benefits and indirect effects must be considered.


Asunto(s)
Cognición , Terapia de Reemplazo Enzimático , Trasplante de Células Madre Hematopoyéticas , Mucopolisacaridosis I/cirugía , Femenino , Humanos , Lactante , Masculino , Estudios Prospectivos , Resultado del Tratamiento
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