Literature DB >> 7983131

Bone-marrow transplantation in Hurler's syndrome. Effect on skeletal development.

R E Field1, J A Buchanan, M G Copplemans, P M Aichroth.   

Abstract

Between 1980 and 1988, displacement bone-marrow transplantation was performed on 25 children with Hurler's syndrome (type-1 mucopolysaccharidosis). We describe the musculoskeletal development of 11 of the 12 surviving children and the orthopaedic procedures undertaken to treat progressive thoracolumbar kyphosis, hip subluxation and carpal tunnel syndrome. We found abnormal bone modelling, focal failures of ossification and an avascular disorder of the femoral head in every patient and offer an explanation for these phenomena. Increasing valgus deformity of the knees and progressive generalised myopathy caused loss of mobility as the children entered adolescence. The benefit of bone-marrow transplantation as a treatment for the skeletal disorders of Hurler's syndrome is limited by the poor penetration of the musculoskeletal tissues by the enzyme derived from the leucocytes.

Entities:  

Mesh:

Year:  1994        PMID: 7983131

Source DB:  PubMed          Journal:  J Bone Joint Surg Br        ISSN: 0301-620X


  47 in total

1.  [Mucopolysaccharidoses].

Authors:  B Link; E Miebach; T Vetter; D Schmitt; M Beck; A Meurer
Journal:  Orthopade       Date:  2008-01       Impact factor: 1.087

2.  Potential role of cathepsin K in the pathophysiology of mucopolysaccharidoses.

Authors:  Susan Wilson; Dieter Brömme
Journal:  J Pediatr Rehabil Med       Date:  2010

Review 3.  Carpal tunnel syndrome: a review.

Authors:  F P Cantatore; F Dell'Accio; G Lapadula
Journal:  Clin Rheumatol       Date:  1997-11       Impact factor: 2.980

4.  Prevalence and development of orthopaedic symptoms in the dutch hurler patient population after haematopoietic stem cell transplantation.

Authors:  F J Stoop; M C Kruyt; M H van der Linden; R J B Sakkers; P M van Hasselt; R M C Castelein
Journal:  JIMD Rep       Date:  2012-09-19

Review 5.  A systematic review of selected musculoskeletal late effects in survivors of childhood cancer.

Authors:  Prasad L Gawade; Melissa M Hudson; Sue C Kaste; Joseph P Neglia; Karen Wasilewski-Masker; Louis S Constine; Leslie L Robison; Kirsten K Ness
Journal:  Curr Pediatr Rev       Date:  2014

Review 6.  Hematopoietic Stem Cell Transplantation for Mucopolysaccharidoses: Past, Present, and Future.

Authors:  Madeleine Taylor; Shaukat Khan; Molly Stapleton; Jianmin Wang; Jing Chen; Robert Wynn; Hiromasa Yabe; Yasutsugu Chinen; Jaap Jan Boelens; Robert W Mason; Francyne Kubaski; Dafne D G Horovitz; Anneliese L Barth; Marta Serafini; Maria Ester Bernardo; Hironori Kobayashi; Kenji E Orii; Yasuyuki Suzuki; Tadao Orii; Shunji Tomatsu
Journal:  Biol Blood Marrow Transplant       Date:  2019-02-14       Impact factor: 5.742

7.  The effect of neonatal gene therapy on skeletal manifestations in mucopolysaccharidosis VII dogs after a decade.

Authors:  Elizabeth M Xing; Van W Knox; Patricia A O'Donnell; Tracey Sikura; Yuli Liu; Susan Wu; Margret L Casal; Mark E Haskins; Katherine P Ponder
Journal:  Mol Genet Metab       Date:  2013-04-06       Impact factor: 4.797

8.  The Kathryn O. and Alan C. Greenberg Center for Skeletal Dysplasias: an interdisciplinary approach.

Authors:  Erin M Carter; Lorraine Montuori; Jessica G Davis; Cathleen L Raggio
Journal:  HSS J       Date:  2008-05-28

9.  Health-related quality of life measures in genetic disorders: an outcome variable for consideration in clinical trials.

Authors:  David A Stevenson; John C Carey
Journal:  Am J Med Genet C Semin Med Genet       Date:  2009-08-15       Impact factor: 3.908

10.  alpha-L-iduronidase therapy for mucopolysaccharidosis type I.

Authors:  Jakub Tolar; Paul J Orchard
Journal:  Biologics       Date:  2008-12
View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.