Replacement therapy with imiglucerase for type 1 Gaucher's disease
Gaucher's disease, the most common sphingolipidosis, is caused by deficiency of the lysosomal enzyme glucocerebrosidase. Therapy with alglucerase (the placental enzyme) is safe and effective at various dosing regimens. We report the use of low-dose imiglucerase (the recombinant enzyme) at two d...
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| Veröffentlicht in: | The Lancet (British edition) 1995-06, Vol.345 (8963), p.1479-1480 |
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| creator | Zimran, A. Elstein, D. Levy-Lahad, E. Zevin, S. Hadas-Halpern, I. Abrahamov, A. Bar-Ziv, Y. Schwartz, A. Foldes, J. |
| description | Gaucher's disease, the most common sphingolipidosis, is caused by deficiency of the lysosomal enzyme glucocerebrosidase. Therapy with alglucerase (the placental enzyme) is safe and effective at various dosing regimens. We report the use of low-dose imiglucerase (the recombinant enzyme) at two dosing schedules: 15 u/kg once fortnightly or 2.5 u/kg thrice weekly. Mean reductions in spleen and liver volumes achieved (in all ten patients) by imiglucerase at 12 months were 36.4% and 14.5%, respectively; mean increase in haemoglobin and platelet counts were 13.4% and 25.7%. There were no serious side-effects. No significant differences were observed between the two schedules. Low-dose low-frequency imiglucerase may be an alternative cost-effective approach with satisfactory clinical response and uncompromised quality of life. |
| doi_str_mv | 10.1016/S0140-6736(95)91038-7 |
| format | Article |
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Therapy with alglucerase (the placental enzyme) is safe and effective at various dosing regimens. We report the use of low-dose imiglucerase (the recombinant enzyme) at two dosing schedules: 15 u/kg once fortnightly or 2.5 u/kg thrice weekly. Mean reductions in spleen and liver volumes achieved (in all ten patients) by imiglucerase at 12 months were 36.4% and 14.5%, respectively; mean increase in haemoglobin and platelet counts were 13.4% and 25.7%. There were no serious side-effects. No significant differences were observed between the two schedules. Low-dose low-frequency imiglucerase may be an alternative cost-effective approach with satisfactory clinical response and uncompromised quality of life.</description><identifier>ISSN: 0140-6736</identifier><identifier>EISSN: 1474-547X</identifier><identifier>DOI: 10.1016/S0140-6736(95)91038-7</identifier><identifier>PMID: 7769903</identifier><identifier>CODEN: LANCAO</identifier><language>eng</language><publisher>London: Elsevier Ltd</publisher><subject>Adolescent ; Adult ; Biological and medical sciences ; Disease ; Drug Administration Schedule ; Drug therapy ; Enzymes ; Errors of metabolism ; Female ; Gaucher Disease - blood ; Gaucher Disease - drug therapy ; Glucosylceramidase - administration & dosage ; Glucosylceramidase - adverse effects ; Humans ; Lipids (lysosomal enzyme disorders, storage diseases) ; Liver - anatomy & histology ; Liver - drug effects ; Male ; Medical research ; Medical sciences ; Metabolic diseases ; Middle Aged ; Quality of life ; Recombinant Proteins - therapeutic use ; Spleen - anatomy & histology ; Spleen - drug effects ; Treatment Outcome</subject><ispartof>The Lancet (British edition), 1995-06, Vol.345 (8963), p.1479-1480</ispartof><rights>1995</rights><rights>1995 INIST-CNRS</rights><rights>Copyright Lancet Ltd. 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Therapy with alglucerase (the placental enzyme) is safe and effective at various dosing regimens. We report the use of low-dose imiglucerase (the recombinant enzyme) at two dosing schedules: 15 u/kg once fortnightly or 2.5 u/kg thrice weekly. Mean reductions in spleen and liver volumes achieved (in all ten patients) by imiglucerase at 12 months were 36.4% and 14.5%, respectively; mean increase in haemoglobin and platelet counts were 13.4% and 25.7%. There were no serious side-effects. No significant differences were observed between the two schedules. Low-dose low-frequency imiglucerase may be an alternative cost-effective approach with satisfactory clinical response and uncompromised quality of life.</description><subject>Adolescent</subject><subject>Adult</subject><subject>Biological and medical sciences</subject><subject>Disease</subject><subject>Drug Administration Schedule</subject><subject>Drug therapy</subject><subject>Enzymes</subject><subject>Errors of metabolism</subject><subject>Female</subject><subject>Gaucher Disease - blood</subject><subject>Gaucher Disease - drug therapy</subject><subject>Glucosylceramidase - administration & dosage</subject><subject>Glucosylceramidase - adverse effects</subject><subject>Humans</subject><subject>Lipids (lysosomal enzyme disorders, storage diseases)</subject><subject>Liver - anatomy & histology</subject><subject>Liver - drug effects</subject><subject>Male</subject><subject>Medical research</subject><subject>Medical sciences</subject><subject>Metabolic diseases</subject><subject>Middle 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Therapy with alglucerase (the placental enzyme) is safe and effective at various dosing regimens. We report the use of low-dose imiglucerase (the recombinant enzyme) at two dosing schedules: 15 u/kg once fortnightly or 2.5 u/kg thrice weekly. Mean reductions in spleen and liver volumes achieved (in all ten patients) by imiglucerase at 12 months were 36.4% and 14.5%, respectively; mean increase in haemoglobin and platelet counts were 13.4% and 25.7%. There were no serious side-effects. No significant differences were observed between the two schedules. Low-dose low-frequency imiglucerase may be an alternative cost-effective approach with satisfactory clinical response and uncompromised quality of life.</abstract><cop>London</cop><pub>Elsevier Ltd</pub><pmid>7769903</pmid><doi>10.1016/S0140-6736(95)91038-7</doi><tpages>2</tpages></addata></record> |
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| subjects | Adolescent Adult Biological and medical sciences Disease Drug Administration Schedule Drug therapy Enzymes Errors of metabolism Female Gaucher Disease - blood Gaucher Disease - drug therapy Glucosylceramidase - administration & dosage Glucosylceramidase - adverse effects Humans Lipids (lysosomal enzyme disorders, storage diseases) Liver - anatomy & histology Liver - drug effects Male Medical research Medical sciences Metabolic diseases Middle Aged Quality of life Recombinant Proteins - therapeutic use Spleen - anatomy & histology Spleen - drug effects Treatment Outcome |
| title | Replacement therapy with imiglucerase for type 1 Gaucher's disease |
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