Literature DB >> 7639557

Behaviour in mucopolysaccharide disorders.

M C Bax1, G A Colville.   

Abstract

This paper reports a study of the nature and prevalence of behaviour problems in 258 children with mucopolysaccharide disorders. Questionnaire data obtained through the post was supplemented by home visits to 42 families in the sample and by regular discussions with families at meetings of the Society for Mucopolysaccharide Diseases. High rates of behaviour problems were found, particularly in children with Sanfilippo's and Hunter's disease aged 5 to 9 years. These included destructiveness, restlessness, and aggressiveness. Sleep problems were common across subtypes with an overall prevalence of 66%. Parents reported that they received little or no support in the management of these difficult behaviours. It is concluded that behaviour problems are a primary feature of the mucopolysaccharide disorders and place a major strain on families. Services to help families cope with these problems are urgently needed.

Entities:  

Mesh:

Year:  1995        PMID: 7639557      PMCID: PMC1511159          DOI: 10.1136/adc.73.1.77

Source DB:  PubMed          Journal:  Arch Dis Child        ISSN: 0003-9888            Impact factor:   3.791


  8 in total

1.  Prevalence of behaviour problems in 3-year-old children: an epidemiological study in a London borough.

Authors:  N Richman; J E Stevenson; P J Graham
Journal:  J Child Psychol Psychiatry       Date:  1975-10       Impact factor: 8.982

Review 2.  The fragile-X syndrome. On the way to a behavioural phenotype.

Authors:  J Turk
Journal:  Br J Psychiatry       Date:  1992-01       Impact factor: 9.319

3.  Adults with Prader-Willi syndrome: a survey of 232 cases.

Authors:  L R Greenswag
Journal:  Dev Med Child Neurol       Date:  1987-04       Impact factor: 5.449

4.  Sleep problems in handicapped children: a preliminary study.

Authors:  J Clements; L Wing; G Dunn
Journal:  J Child Psychol Psychiatry       Date:  1986-05       Impact factor: 8.982

5.  Lesch-Nyhan disease: clinical experience with nineteen patients.

Authors:  R Christie; C Bay; I A Kaufman; B Bakay; M Borden; W L Nyhan
Journal:  Dev Med Child Neurol       Date:  1982-06       Impact factor: 5.449

6.  Developmental and degenerative patterns associated with cognitive, behavioural and motor difficulties in the Sanfilippo syndrome: an epidemiological study.

Authors:  F D Nidiffer; T E Kelly
Journal:  J Ment Defic Res       Date:  1983-09

7.  Genetic heterogeneity and clinical variability in the Sanfilippo syndrome (types A, B, and C).

Authors:  J J van de Kamp; M F Niermeijer; K von Figura; M A Giesberts
Journal:  Clin Genet       Date:  1981-08       Impact factor: 4.438

8.  Behavioral and emotional problems in youngsters with Prader-Willi syndrome.

Authors:  L M Curfs; F C Verhulst; J P Fryns
Journal:  Genet Couns       Date:  1991
  8 in total
  35 in total

1.  Health-Related Quality of Life in Patients with MPS II.

Authors:  Mary Needham; Wendy Packman; Natasha Quinn; Maxwell Rappoport; Christa Aoki; Alan Bostrom; Matthew Cordova; Sandra Macias; Cynthia Morgan; Seymour Packman
Journal:  J Genet Couns       Date:  2014-11-14       Impact factor: 2.537

2.  Sleep-related breathing in children with mucopolysaccharidosis.

Authors:  A Nashed; S Al-Saleh; J Gibbons; I MacLusky; J MacFarlane; A Riekstins; J Clarke; I Narang
Journal:  J Inherit Metab Dis       Date:  2009-06-28       Impact factor: 4.982

3.  Sleep disturbance in Sanfilippo syndrome: a parental questionnaire study.

Authors:  J Fraser; A A Gason; J E Wraith; M B Delatycki
Journal:  Arch Dis Child       Date:  2005-12       Impact factor: 3.791

4.  Psychopharmacological treatment of behavioural problems in Sanfilippo syndrome.

Authors:  Paramala Santosh; Jennifer Ruth Parker; Lynn Jones
Journal:  BMJ Case Rep       Date:  2009-03-17

5.  Social Functioning and Behaviour in Mucopolysaccharidosis IH [Hurlers Syndrome].

Authors:  Annukka Lehtonen; Stewart Rust; Simon Jones; Richard Brown; Dougal Hare
Journal:  JIMD Rep       Date:  2017-07-29

6.  Coping Strategies, Stress, and Support Needs in Caregivers of Children with Mucopolysaccharidosis.

Authors:  Amy Schadewald; Ericka Kimball; Li Ou
Journal:  JIMD Rep       Date:  2018-01-04

7.  Pain: a prevalent feature in patients with mucopolysaccharidosis. Results of a cross-sectional national survey.

Authors:  Marion M G Brands; Deniz Güngör; Johanna M P van den Hout; Francois P J Karstens; Esmee Oussoren; Iris Plug; Jaap Jan Boelens; Peter M van Hasselt; Carla E M Hollak; Margot F Mulder; Estela Rubio Gozalbo; Jan A Smeitink; G Peter A Smit; Frits A Wijburg; Hanka Meutgeert; Ans T van der Ploeg
Journal:  J Inherit Metab Dis       Date:  2014-07-22       Impact factor: 4.982

Review 8.  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

9.  Parental social support, coping strategies, resilience factors, stress, anxiety and depression levels in parents of children with MPS III (Sanfilippo syndrome) or children with intellectual disabilities (ID).

Authors:  Sheena Grant; Elaine Cross; James Edmond Wraith; Simon Jones; Louise Mahon; Michelle Lomax; Brian Bigger; Dougal Hare
Journal:  J Inherit Metab Dis       Date:  2012-11-15       Impact factor: 4.982

10.  MPS II: adaptive behavior of patients and impact on the family system.

Authors:  Mary Needham; Wendy Packman; Maxwell Rappoport; Natasha Quinn; Matthew Cordova; Sandra Macias; Cynthia Morgan; Seymour Packman
Journal:  J Genet Couns       Date:  2013-11-06       Impact factor: 2.537

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.