Literature DB >> 18278070

Growth and endocrine function in patients with Hurler syndrome after hematopoietic stem cell transplantation.

L E Polgreen1, J Tolar, M Plog, J H Himes, P J Orchard, C B Whitley, B S Miller, A Petryk.   

Abstract

Short stature is characteristic of Hurler syndrome, or mucopolysaccharidosis type IH (MPS IH). Hematopoietic stem cell transplantation (HSCT) is used to treat children with MPS IH. While HSCT corrects some of the metabolic features of MPS IH, its effects on growth are not well delineated. We investigated growth in patients with MPS IH after HSCT and described accompanying endocrine abnormalities. A cohort of 48 patients with MPS IH who had received HSCT between 1983 and 2005 were included. The prevalence of short stature (height <-2 s.d. score, SDS) before HSCT was 9%, and increased to 71% at last follow-up (6.9+/-5.1 years after HSCT). Short stature was positively associated with increased age at HSCT (P=0.002) and TBI (P=0.009). In total, 23% had growth hormone deficiency and/or low insulin-like growth factor-1, one female patient had premature adrenarche, one precocious puberty and 27% had clinical or subclinical hypothyroidism. Growth failure is highly prevalent in children with MPS IH after HSCT. Children who had no TBI exposure and were younger at the time of HSCT had a better height outcome.

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Mesh:

Year:  2008        PMID: 18278070     DOI: 10.1038/bmt.2008.20

Source DB:  PubMed          Journal:  Bone Marrow Transplant        ISSN: 0268-3369            Impact factor:   5.483


  34 in total

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Authors:  J de Ruijter; L Broere; M F Mulder; A T van der Ploeg; M E Rubio-Gozalbo; S B Wortmann; G Visser; F A Wijburg
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2.  Replacing the enzyme alpha-L-iduronidase at birth ameliorates symptoms in the brain and periphery of dogs with mucopolysaccharidosis type I.

Authors:  Ashley D Dierenfeld; Michael F McEntee; Carole A Vogler; Charles H Vite; Agnes H Chen; Merry Passage; Steven Le; Sahil Shah; Jackie K Jens; Elizabeth M Snella; Karen L Kline; Jennifer D Parkes; Wendy A Ware; Lori E Moran; Amanda J Fales-Williams; Jane A Wengert; R David Whitley; Daniel M Betts; Amy M Boal; Elizabeth A Riedesel; William Gross; N Matthew Ellinwood; Patricia I Dickson
Journal:  Sci Transl Med       Date:  2010-12-01       Impact factor: 17.956

3.  A mouse model suggests two mechanisms for thyroid alterations in infantile cystinosis: decreased thyroglobulin synthesis due to endoplasmic reticulum stress/unfolded protein response and impaired lysosomal processing.

Authors:  H P Gaide Chevronnay; V Janssens; P Van Der Smissen; X H Liao; Y Abid; N Nevo; C Antignac; S Refetoff; S Cherqui; C E Pierreux; P J Courtoy
Journal:  Endocrinology       Date:  2015-03-26       Impact factor: 4.736

4.  Elevated TNF-α is associated with pain and physical disability in mucopolysaccharidosis types I, II, and VI.

Authors:  Lynda E Polgreen; Richard K Vehe; Kyle Rudser; Alicia Kunin-Batson; Jeanine Jarnes Utz; Patricia Dickson; Elsa Shapiro; Chester B Whitley
Journal:  Mol Genet Metab       Date:  2016-01-28       Impact factor: 4.797

5.  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

6.  Combined spinal arthrodesis with instrumentation for the management of progressive thoracolumbar kyphosis in children with mucopolysaccharidosis.

Authors:  Enrique Garrido; Félix Tomé-Bermejo; Christopher I Adams
Journal:  Eur Spine J       Date:  2014-01-28       Impact factor: 3.134

7.  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

8.  Four successful pregnancies in a patient with mucopolysaccharidosis type I treated by allogeneic bone marrow transplantation.

Authors:  G Remérand; E Merlin; R Froissart; F Brugnon; J Kanold; L Janny; F Deméocq
Journal:  J Inherit Metab Dis       Date:  2009-03-13       Impact factor: 4.982

9.  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

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

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