Literature DB >> 1907534

Hurler syndrome with special reference to histologic abnormalities of the growth plate.

C P Silveri1, F S Kaplan, M D Fallon, E Bayever, C S August.   

Abstract

Hurler syndrome is a mucopolysaccharide disorder resulting from an heritable deficiency in alpha-L-iduronidase, an enzyme required in the catabolism of heparan sulfate and dermatan sulfate glycosaminoglycan (GAGs). The resultant intracellular accumulation of GAG leads to disruption of the intracellular and extracellular environment and dysfunction of multiple organ systems. Among the most noted manifestations of this disease is disproportionate short trunk dwarfism, which develops during the first years of life. Histochemical and electron-microscopic observations on a 30-month-old child with Hurler syndrome showed marked irregularities in chondrocyte orientation within the growth plate, along with disruption of the normal columnar architecture. Vacuolization with enlargement of the cellular border was the characteristic ultrastructural finding. An heritable abnormality in the enzymatic degradation of structural glycosaminoglycans leads to profound disruption of the normal mechanisms of growth and development.

Entities:  

Mesh:

Year:  1991        PMID: 1907534

Source DB:  PubMed          Journal:  Clin Orthop Relat Res        ISSN: 0009-921X            Impact factor:   4.176


  18 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

3.  Growth patterns and the use of growth hormone in the mucopolysaccharidoses.

Authors:  L E Polgreen; B S Miller
Journal:  J Pediatr Rehabil Med       Date:  2010

4.  Biomarkers of bone remodeling in children with mucopolysaccharidosis types I, II, and VI.

Authors:  David A Stevenson; Kyle Rudser; Alicia Kunin-Batson; Ellen B Fung; David Viskochil; Elsa Shapiro; Paul J Orchard; Chester B Whitley; Lynda E Polgreen
Journal:  J Pediatr Rehabil Med       Date:  2014

5.  Effect of recombinant human growth hormone on changes in height, bone mineral density, and body composition over 1-2 years in children with Hurler or Hunter syndrome.

Authors:  Lynda E Polgreen; William Thomas; Paul J Orchard; Chester B Whitley; Bradley S Miller
Journal:  Mol Genet Metab       Date:  2013-12-11       Impact factor: 4.797

6.  Surgical management of thoracolumbar kyphosis in mucopolysaccharidosis type 1 in a reference center.

Authors:  Kariman Abelin Genevois; Christophe Garin; Federico Solla; Nathalie Guffon; Rémi Kohler
Journal:  J Inherit Metab Dis       Date:  2013-06-29       Impact factor: 4.982

Review 7.  Mucopolysaccharidoses.

Authors:  Rolando Cimaz; Francesco La Torre
Journal:  Curr Rheumatol Rep       Date:  2014-01       Impact factor: 4.592

8.  Low bone mineral content and challenges in interpretation of dual-energy X-ray absorptiometry in children with mucopolysaccharidosis types I, II, and VI.

Authors:  Lynda E Polgreen; William Thomas; Ellen Fung; David Viskochil; David A Stevenson; Julia Steinberger; Paul Orchard; Chester B Whitley; Kristine E Ensrud
Journal:  J Clin Densitom       Date:  2013-04-02       Impact factor: 2.617

Review 9.  Failures of Endochondral Ossification in the Mucopolysaccharidoses.

Authors:  Zhirui Jiang; Sharon Byers; Margret L Casal; Lachlan J Smith
Journal:  Curr Osteoporos Rep       Date:  2020-10-16       Impact factor: 5.096

10.  Short-term growth hormone treatment in children with Hurler syndrome after hematopoietic cell transplantation.

Authors:  L E Polgreen; M Plog; J D Schwender; J Tolar; W Thomas; P J Orchard; B S Miller; A Petryk
Journal:  Bone Marrow Transplant       Date:  2009-03-02       Impact factor: 5.483

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