Literature DB >> 8640962

Early presentation in the mucopolysaccharide disorders.

G A Colville1, M A Bax.   

Abstract

The findings of an international questionnaire study of 258 children, affected by the four main subtypes of mucopolysaccharidosis, are presented. Questionnaires were completed by a parent or main carer and all subjects were alive at the time of contact and suffering from Hurler, Hunter, Sanfilippo or Morquio syndrome. A significant proportion of parents of Hurler children (24%) were unaware that anything was wrong with their baby before diagnosis but a larger number (45%) had felt concerned about their child's appearance. Similarly, in the case of the Morquio children, in 75% of cases, parents had been worried about some aspect of their child's physical appearance. In contrast, it was frequently delayed or regressing language which alerted parents of Sanfilippo (56%) and Hunter (32%) children, and this was associated with behaviour problems in 43% of Sanfilippo cases. There were many cases of delayed diagnosis, often occurring more than 2 years after concerns were first raised.

Entities:  

Mesh:

Year:  1996        PMID: 8640962

Source DB:  PubMed          Journal:  Child Care Health Dev        ISSN: 0305-1862            Impact factor:   2.508


  8 in total

1.  Mucopolysaccharidosis Type IIIA presents as a variant of Klüver-Bucy syndrome.

Authors:  Michael Potegal; Brianna Yund; Kyle Rudser; Alia Ahmed; Kate Delaney; Igor Nestrasil; Chester B Whitley; Elsa G Shapiro
Journal:  J Clin Exp Neuropsychol       Date:  2013-06-08       Impact factor: 2.475

2.  Quantifying behaviors of children with Sanfilippo syndrome: the Sanfilippo Behavior Rating Scale.

Authors:  Elsa G Shapiro; Igor Nestrasil; Alia Ahmed; Andrew Wey; Kyle R Rudser; Kathleen A Delaney; Robin K Rumsey; Patrick A J Haslett; Chester B Whitley; Michael Potegal
Journal:  Mol Genet Metab       Date:  2015-03-05       Impact factor: 4.797

Review 3.  Behavioural phenotypes of the mucopolysaccharide disorders: a systematic literature review of cognitive, motor, social, linguistic and behavioural presentation in the MPS disorders.

Authors:  E M Cross; D J Hare
Journal:  J Inherit Metab Dis       Date:  2013-02-06       Impact factor: 4.982

4.  Diagnosis and treatment trends in mucopolysaccharidosis I: findings from the MPS I Registry.

Authors:  Kristin D'Aco; Lisa Underhill; Lakshmi Rangachari; Pamela Arn; Gerald F Cox; Roberto Giugliani; Torayuki Okuyama; Frits Wijburg; Paige Kaplan
Journal:  Eur J Pediatr       Date:  2012-01-11       Impact factor: 3.183

5.  An algorithm to predict phenotypic severity in mucopolysaccharidosis type I in the first month of life.

Authors:  Sandra D K Kingma; Eveline J Langereis; Clasine M de Klerk; Lida Zoetekouw; Tom Wagemans; Lodewijk IJlst; Ronald J A Wanders; Frits A Wijburg; Naomi van Vlies
Journal:  Orphanet J Rare Dis       Date:  2013-07-09       Impact factor: 4.123

6.  Early disease progression of Hurler syndrome.

Authors:  Bridget T Kiely; Jennifer L Kohler; Hannah Y Coletti; Michele D Poe; Maria L Escolar
Journal:  Orphanet J Rare Dis       Date:  2017-02-14       Impact factor: 4.123

7.  Unresolving short stature in a possible case of mucopolysccharidosis.

Authors:  Ac Ayuk; Ho Obu; Md Ughasoro; Ns Ibeziako
Journal:  Ann Med Health Sci Res       Date:  2014-03

Review 8.  Mucopolysaccharidosis Type I: A Review of the Natural History and Molecular Pathology.

Authors:  Christiane S Hampe; Julie B Eisengart; Troy C Lund; Paul J Orchard; Monika Swietlicka; Jacob Wesley; R Scott McIvor
Journal:  Cells       Date:  2020-08-05       Impact factor: 6.600

  8 in total

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