American journal of human genetics · 2009
Use of a modified alpha-N-acetylgalactosaminidase in the development of enzyme replacement therapy for Fabry disease.
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Tajima Youichi, Kawashima Ikuo, Tsukimura Takahiro, Sugawara Kanako, Kuroda Mayuko, Suzuki Toshihiro, Togawa Tadayasu, Chiba Yasunori, Jigami Yoshifumi, Ohno Kazuki, Fukushige Tomoko, Kanekura Takuro, Itoh Kohji, Ohashi Toya, Sakuraba Hitoshi
Аннотация
A modified alpha-N-acetylgalactosaminidase (NAGA) with alpha-galactosidase A (GLA)-like substrate specificity was designed on the basis of structural studies and was produced in Chinese hamster ovary cells. The enzyme acquired the ability to catalyze the degradation of 4-methylumbelliferyl-alpha-D-galactopyranoside. It retained the original NAGA's stability in plasma and N-glycans containing many mannose 6-phosphate (M6P) residues, which are advantageous for uptake by cells via M6P receptors. There was no immunological cross-reactivity between the modified NAGA and GLA, and the modified NAGA did not react to serum from a patient with Fabry disease recurrently treated with a recombinant GLA. The enzyme cleaved globotriaosylceramide (Gb3) accumulated in cultured fibroblasts from a patient with Fabry disease. Furthermore, like recombinant GLA proteins presently used for enzyme replacement therapy (ERT) for Fabry disease, the enzyme intravenously injected into Fabry model mice prevented Gb3 storage in the liver, kidneys, and heart and improved the pathological changes in these organs. Because this modified NAGA is hardly expected to cause an allergic reaction in Fabry disease patients, it is highly promising as a new and safe enzyme for ERT for Fabry disease.
Предметные рубрики MeSH
Amino Acid SubstitutionAnimalsBinding SitesCHO CellsCatalysisCells, CulturedCricetinaeCricetulusCulture Media, ConditionedDNA, ComplementaryDisease Models, AnimalDrug StabilityEnzyme Replacement TherapyFabry DiseaseFibroblastsFluorescent DyesGalactosidesGenetic VectorsHumansHydrogen-Ion ConcentrationHymecromoneImmunohistochemistryKidneyLiverMiceMice, KnockoutModels, MolecularMolecular WeightMyocardiumRecombinant ProteinsRetroviridaeTransfectionTrihexosylceramidesalpha-N-Acetylgalactosaminidase