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Supraphysiological plasma levels of IDUAe, not IDUAc, lead to behavioral normalization of MPS I mice in repeated open-field test. Age-matched MPS I, normal heterozygote (HET) or gene therapy treated mice were evaluated 6–7 months after transplantation for changes in (a) horizontal activities and (b) grooming activities. Data are presented as mean ± SEM. *P < 0.05. IDUA, α-l-iduronidase; MPS I, mucopolysaccharidosis type I.
