Short-Term Efficacy of Enzyme Replacement Therapy in Korean Patients with Fabry Disease
Identifieur interne : 002C80 ( Ncbi/Curation ); précédent : 002C79; suivant : 002C81Short-Term Efficacy of Enzyme Replacement Therapy in Korean Patients with Fabry Disease
Auteurs : Jin-Ho Choi [Corée du Sud] ; Young Mi Cho [Corée du Sud] ; Kwang-Sun Suh [Corée du Sud] ; Hye-Ran Yoon [Corée du Sud] ; Gu-Hwan Kim [Corée du Sud] ; Sung-Su Kim [Corée du Sud] ; Jung Min Ko [Corée du Sud] ; Joo Hoon Lee [Corée du Sud] ; Young Seo Park [Corée du Sud] ; Han-Wook Yoo [Corée du Sud]Source :
- Journal of Korean Medical Science [ 1011-8934 ] ; 2008.
Abstract
Fabrazyme has been widely used for treatment of Fabry disease since its approval by the U.S. Food and Drug Administration in 2003. This study was undertaken to assess the short-term efficacy and safety of enzyme replacement therapy (ERT) for Fabry disease in Korea. Eight male patients and three female symptomatic carriers aged 13 to 48 yr were included. Fabrazyme was administered by intravenous infusion at a dose of 1 mg/kg every 2 weeks. Plasma and urine globotriaosylceramide (GL-3) levels, serum creatinine, creatinine clearance, and 24-hr urine protein levels were measured every 3 months. Kidney biopsies, ophthalmologic exams, and pure tone audiometry were performed before and 1 yr after ERT. Kidney function, including serum creatinine, creatinine clearance, and the 24-hr urine protein level, remained stable during ERT. Plasma and urine GL-3 levels were reduced within 3 to 6 months of ERT initiation. Microvascular endothelial deposits of GL-3 were decreased from renal biopsy specimens after 1 yr of treatment. The severity of sensorineural hearing loss and tinnitus did not improve after ERT. ERT is safe and effective in stabilizing renal function and clearing microvascular endothelial GL-3 from kidney biopsy specimen in Korean patients with Fabry disease.
Url:
DOI: 10.3346/jkms.2008.23.2.243
PubMed: 18437007
PubMed Central: 2526436
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sort="Choi, Jin Ho" uniqKey="Choi J" first="Jin-Ho" last="Choi">Jin-Ho Choi</name><affiliation wicri:level="1"><nlm:aff id="A1">Department of Pediatrics, Research Institute for Medical Sciences, Chungnam National University Hospital, College of Medicine, Chungnam National University, Daejeon, Korea.</nlm:aff><country xml:lang="fr" wicri:curation="lc">Corée du Sud</country><wicri:regionArea>Department of Pediatrics, Research Institute for Medical Sciences, Chungnam National University Hospital, College of Medicine, Chungnam National University, Daejeon</wicri:regionArea><wicri:noRegion>Daejeon</wicri:noRegion></affiliation></author><author><name sortKey="Cho, Young Mi" sort="Cho, Young Mi" uniqKey="Cho Y" first="Young Mi" last="Cho">Young Mi Cho</name><affiliation wicri:level="1"><nlm:aff id="A2">Department of Pathology, Research Institute for Medical Sciences, Chungnam National University Hospital, College of Medicine, Chungnam National University, Daejeon, Korea.</nlm:aff><country xml:lang="fr" wicri:curation="lc">Corée du Sud</country><wicri:regionArea>Department of Pathology, Research Institute for Medical Sciences, Chungnam National University Hospital, College of Medicine, Chungnam National University, Daejeon</wicri:regionArea><wicri:noRegion>Daejeon</wicri:noRegion></affiliation></author><author><name sortKey="Suh, Kwang Sun" sort="Suh, Kwang Sun" uniqKey="Suh K" first="Kwang-Sun" last="Suh">Kwang-Sun Suh</name><affiliation wicri:level="3"><nlm:aff id="A3">Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea.</nlm:aff><country xml:lang="fr" wicri:curation="lc">Corée du Sud</country><wicri:regionArea>Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul</wicri:regionArea><placeName><settlement type="city">Séoul</settlement><region type="capital">Région capitale de Séoul</region></placeName></affiliation></author><author><name sortKey="Yoon, Hye Ran" sort="Yoon, Hye Ran" 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University of Ulsan College of Medicine, Seoul</wicri:regionArea><placeName><settlement type="city">Séoul</settlement><region type="capital">Région capitale de Séoul</region></placeName></affiliation></author><author><name sortKey="Kim, Sung Su" sort="Kim, Sung Su" uniqKey="Kim S" first="Sung-Su" last="Kim">Sung-Su Kim</name><affiliation wicri:level="3"><nlm:aff id="A5">Department of Pediatrics, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea.</nlm:aff><country xml:lang="fr" wicri:curation="lc">Corée du Sud</country><wicri:regionArea>Department of Pediatrics, Asan Medical Center, University of Ulsan College of Medicine, Seoul</wicri:regionArea><placeName><settlement type="city">Séoul</settlement><region type="capital">Région capitale de Séoul</region></placeName></affiliation></author><author><name sortKey="Ko, Jung Min" sort="Ko, Jung Min" uniqKey="Ko J" first="Jung Min" last="Ko">Jung Min Ko</name><affiliation wicri:level="3"><nlm:aff id="A6">Biomedical and Pharmaceutical Analysis Laboratory, Department of Analytical Chemistry, School of Pharmacy, Duksung Women's University, Seoul, Korea.</nlm:aff><country xml:lang="fr" wicri:curation="lc">Corée du Sud</country><wicri:regionArea>Biomedical and Pharmaceutical Analysis Laboratory, Department of Analytical Chemistry, School of Pharmacy, Duksung Women's University, Seoul</wicri:regionArea><placeName><settlement type="city">Séoul</settlement><region type="capital">Région capitale de Séoul</region></placeName></affiliation></author><author><name sortKey="Lee, Joo Hoon" sort="Lee, Joo Hoon" uniqKey="Lee J" first="Joo Hoon" last="Lee">Joo Hoon Lee</name><affiliation wicri:level="3"><nlm:aff id="A6">Biomedical and Pharmaceutical Analysis Laboratory, Department of Analytical Chemistry, School of Pharmacy, Duksung Women's University, Seoul, Korea.</nlm:aff><country xml:lang="fr" wicri:curation="lc">Corée du Sud</country><wicri:regionArea>Biomedical and Pharmaceutical Analysis Laboratory, Department 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sort="Yoo, Han Wook" uniqKey="Yoo H" first="Han-Wook" last="Yoo">Han-Wook Yoo</name><affiliation wicri:level="3"><nlm:aff id="A6">Biomedical and Pharmaceutical Analysis Laboratory, Department of Analytical Chemistry, School of Pharmacy, Duksung Women's University, Seoul, Korea.</nlm:aff><country xml:lang="fr" wicri:curation="lc">Corée du Sud</country><wicri:regionArea>Biomedical and Pharmaceutical Analysis Laboratory, Department of Analytical Chemistry, School of Pharmacy, Duksung Women's University, Seoul</wicri:regionArea><placeName><settlement type="city">Séoul</settlement><region type="capital">Région capitale de Séoul</region></placeName></affiliation></author></analytic><series><title level="j">Journal of Korean Medical Science</title><idno type="ISSN">1011-8934</idno><idno type="eISSN">1598-6357</idno><imprint><date when="2008">2008</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Fabrazyme has been widely used for treatment of Fabry disease since its approval by the U.S. Food and Drug Administration in 2003. This study was undertaken to assess the short-term efficacy and safety of enzyme replacement therapy (ERT) for Fabry disease in Korea. Eight male patients and three female symptomatic carriers aged 13 to 48 yr were included. Fabrazyme was administered by intravenous infusion at a dose of 1 mg/kg every 2 weeks. Plasma and urine globotriaosylceramide (GL-3) levels, serum creatinine, creatinine clearance, and 24-hr urine protein levels were measured every 3 months. Kidney biopsies, ophthalmologic exams, and pure tone audiometry were performed before and 1 yr after ERT. Kidney function, including serum creatinine, creatinine clearance, and the 24-hr urine protein level, remained stable during ERT. Plasma and urine GL-3 levels were reduced within 3 to 6 months of ERT initiation. Microvascular endothelial deposits of GL-3 were decreased from renal biopsy specimens after 1 yr of treatment. The severity of sensorineural hearing loss and tinnitus did not improve after ERT. ERT is safe and effective in stabilizing renal function and clearing microvascular endothelial GL-3 from kidney biopsy specimen in Korean patients with Fabry disease.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Kint, Ja" uniqKey="Kint J">JA Kint</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Desnick, Rj" uniqKey="Desnick R">RJ Desnick</name></author><author><name sortKey="Ioannou, Ya" uniqKey="Ioannou Y">YA Ioannou</name></author><author><name sortKey="Eng, Cm" uniqKey="Eng C">CM Eng</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Sheth, Kj" uniqKey="Sheth K">KJ Sheth</name></author><author><name sortKey="Roth, Da" uniqKey="Roth D">DA Roth</name></author><author><name sortKey="Adams, Mb" uniqKey="Adams M">MB Adams</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Peters, Fp" uniqKey="Peters F">FP 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Keilmann</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Wilcox, Wr" uniqKey="Wilcox W">WR Wilcox</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Mignani, R" uniqKey="Mignani R">R Mignani</name></author><author><name sortKey="Panichi, V" uniqKey="Panichi V">V Panichi</name></author><author><name sortKey="Giudicissi, A" uniqKey="Giudicissi A">A Giudicissi</name></author><author><name sortKey="Taccola, D" uniqKey="Taccola D">D Taccola</name></author><author><name sortKey="Boscaro, F" uniqKey="Boscaro F">F Boscaro</name></author><author><name sortKey="Feletti, C" uniqKey="Feletti C">C Feletti</name></author><author><name sortKey="Moneti, G" uniqKey="Moneti G">G Moneti</name></author><author><name sortKey="Cagnoli, L" uniqKey="Cagnoli L">L Cagnoli</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Brooks, Da" uniqKey="Brooks D">DA Brooks</name></author><author><name sortKey="Kakavanos, R" uniqKey="Kakavanos R">R Kakavanos</name></author><author><name sortKey="Hopwood, Jj" uniqKey="Hopwood J">JJ Hopwood</name></author></analytic></biblStruct></listBibl></div1></back></TEI></record>Pour manipuler ce document sous Unix (Dilib)
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