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• W2000654140 abstract "Bone marrow transplantation has been used for >20 years to alter the lethal natural history of Hurler disease, a progressive systemic disease in which glycosaminoglycans accumulate in all body tissues due to the absence of the lysosomal enzyme α-L-iduronidase. 1 Hobbs J. Hugh-Jones K. Barrett A. Byrom N. Chambers D. Henry K. James D. Lucas C. Robes T. Benson P. et al. Reversal of clinical features of Hurler’s disease and biochemical improvement after treatment by bone marrow transplantation. Lancet. 1981; 2: 709-712 Abstract PubMed Scopus (385) Google Scholar , 2 Krivit W. Shapiro E.G. Balthazor M. Lockman L.A. Summers C.G. House J.J. Ogilivie J. Whitley C.B. Belani K.G. Braunlin E. et al. Hurler syndrome outcomes and planning following bone marrow transplantation. in: Steward C.G. Hobbs J.R. Correction of Genetic Disease by Transplantation, III. COGENT, London1995: 25-40 Google Scholar Deposition of glycosaminoglycans within the myointima of the epicardial coronary arteries causes heart failure and is responsible for death within the first decade of life. 3 Gelb B.D. Metabolic heart disease. in: Garson Jr, A. Bricker J.T. McNamara D.G. The Science and Practice of Pediatric Cardiology. Lea & Febiger, Philadelphia, PA1990: 1656-1683 Google Scholar , 4 Krovetz L.J. Schiebler G.L. Cardiovascular manifestations of the genetic mucopolysaccharidoses. Birth Defects. 1981; 8: 192-196 Google Scholar , 5 Brosius F.C. Roberts W.C. Coronary artery disease in the Hurler syndrome. Am J Cardiol. 1981; 47: 649-653 Abstract Full Text PDF PubMed Scopus (50) Google Scholar Successful engraftment of bone marrow ameliorates the disease process within the liver, the large airways, and the central nervous system. 2 Krivit W. Shapiro E.G. Balthazor M. Lockman L.A. Summers C.G. House J.J. Ogilivie J. Whitley C.B. Belani K.G. Braunlin E. et al. Hurler syndrome outcomes and planning following bone marrow transplantation. in: Steward C.G. Hobbs J.R. Correction of Genetic Disease by Transplantation, III. COGENT, London1995: 25-40 Google Scholar Normalization of α-L-iduronidase levels is responsible for these salutary effects. 6 Whitley C.B. Belani K.G. Chang P.N. Summers C.G. Blazar B.R. Tsai M.Y. Latchaw R.E. Ramsay N.K. Kersey J.H. Long-term outcome of Hurler syndrome following bone marrow transplantation. Am J Med Genet. 1981; 46: 209-218 Crossref Scopus (171) Google Scholar The clinical cardiac status of patients with Hurler disease after successful engraftment is significantly different from historical controls as evidenced by prolonged survival. 7 Krivit W. Henslee-Downley J. Klemperer M. Cown M. Peters C. Sanders J. Saunders F. Weinstein H. Williams T. Harris R. et al. Survival in Hurler’s disease following bone marrow transplantation in 84 patients. Transplantation. 1981; 15: S182-S185 Google Scholar To date, pathologic evidence has been unavailable to document the effect of successful engraftment within the epicardial coronary arteries of persons with Hurler syndrome." @default.
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• W2000654140 title "Coronary artery patency following long-term successful engraftment 14 years after bone marrow transplantation in the hurler syndrome" @default.
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