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

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.

journal_name

Bone Marrow Transplant

journal_title

Bone marrow transplantation

authors

Polgreen LE,Tolar J,Plog M,Himes JH,Orchard PJ,Whitley CB,Miller BS,Petryk A

doi

10.1038/bmt.2008.20

subject

Has Abstract

pub_date

2008-06-01 00:00:00

pages

1005-11

issue

12

eissn

0268-3369

issn

1476-5365

pii

bmt200820

journal_volume

41

pub_type

杂志文章

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