Literature DB >> 6377066

Bone-marrow transplantation in severe Gaucher's disease.

J M Rappeport, E I Ginns.   

Abstract

We performed allogeneic bone-marrow transplantation of normal cells in an eight-year-old patient with Type 3 Gaucher's disease in an attempt to alter his progressive deterioration. The procedure resulted in complete engraftment of the enzymatically normal donor cells. Donor monocyte precursors were present in the circulation of the recipient by 28 days after transplantation. Post-transplantation beta-glucocerebrosidase activity in mononuclear white cells in peripheral blood exceeded normal levels. Despite these early results no change in Gaucher's-cell infiltration of the bone marrow was noted for at least 111 days, and Gaucher's cells persisted in the marrow for at least 274 days. The plasma glucocerebroside concentration ultimately returned to normal. During the post-transplantation course, until the patient died from an episode of sepsis, there was no important change in his clinical status. However, this case demonstrates that the enzymatic abnormality in hematopoietic cells in Gaucher's disease is correctable by bone-marrow transplantation, leading to normalization of plasma levels of glucocerebroside. We observed the long survival of Gaucher's cells, which was consistent with the lack of clinical improvement. Intervention might have been more effective earlier in the course of the disease.

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Year:  1984        PMID: 6377066     DOI: 10.1056/NEJM198407123110203

Source DB:  PubMed          Journal:  N Engl J Med        ISSN: 0028-4793            Impact factor:   91.245


  18 in total

Review 1.  Enzyme-replacement therapy: problems and prospects.

Authors:  B Rademaker; J Raber
Journal:  Pharm Weekbl Sci       Date:  1989-10-20

2.  Production of human glucocerebrosidase in mice after retroviral gene transfer into multipotential hematopoietic progenitor cells.

Authors:  P H Correll; J K Fink; R O Brady; L K Perry; S Karlsson
Journal:  Proc Natl Acad Sci U S A       Date:  1989-11       Impact factor: 11.205

Review 3.  Alglucerase. A pharmacoeconomic appraisal of its use in the treatment of Gaucher's disease.

Authors:  R Whittington; K L Goa
Journal:  Pharmacoeconomics       Date:  1995-01       Impact factor: 4.981

Review 4.  Gaucher disease as a paradigm of current issues regarding single gene mutations of humans.

Authors:  E Beutler
Journal:  Proc Natl Acad Sci U S A       Date:  1993-06-15       Impact factor: 11.205

Review 5.  Hematopoietic stem cell transplantation for Gaucher disease.

Authors:  Usha R Somaraju; Krishna Tadepalli
Journal:  Cochrane Database Syst Rev       Date:  2017-10-18

Review 6.  Transplantation as disease modifying therapy in adults with inherited metabolic disorders.

Authors:  Sandra Sirrs; Fady Hannah-Shmouni; Stephen Nantel; James Neuberger; Eric M Yoshida
Journal:  J Inherit Metab Dis       Date:  2018-02-01       Impact factor: 4.982

7.  Phenotypic and genotypic heterogeneity in gaucher disease: Implications for genetic counseling.

Authors:  E Sidransky; E I Ginns
Journal:  J Genet Couns       Date:  1994-03       Impact factor: 2.537

8.  Complete correction of the enzymatic defect of type I Gaucher disease fibroblasts by retroviral-mediated gene transfer.

Authors:  J Sorge; W Kuhl; C West; E Beutler
Journal:  Proc Natl Acad Sci U S A       Date:  1987-02       Impact factor: 11.205

9.  Failure to demonstrate pluripotential hemopoietic stem cells in mouse brains.

Authors:  P M Hoogerbrugge; G Wagemaker; D W van Bekkum
Journal:  Proc Natl Acad Sci U S A       Date:  1985-06       Impact factor: 11.205

10.  Musings on genome medicine: enzyme-replacement therapy of the lysosomal storage diseases.

Authors:  David G Nathan; Stuart H Orkin
Journal:  Genome Med       Date:  2009-12-09       Impact factor: 11.117

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