Literature DB >> 22447152

Living situation, occupation and health-related quality of life in adult patients with classic galactosemia.

Björn Hoffmann1, Nico Dragano, Susanne Schweitzer-Krantz.   

Abstract

BACKGROUND: Galactose-1-phosphate uridyltransferase deficiency is well known as the underlying defect in classic galactosemia. However, little is known about the consequences of this defect beyond physical disease. AIM: To evaluate psychosocial, educational and occupational outcome as well as health-related quality of life (HRQOL) in adult German patients with galactosemia and to compare information with data from patients with phenylketonuria as well as the general German population.
METHODS: Members of the German patient support group for galactosemia received invitation, informed consent form and questionnaires by regular mail from the patient support group. Participation was voluntary.
RESULTS: Forty-one out of 66 invited patients participated in this study. Nearly 2/3 of the patients were singles, and the majority of patients were still living with their parents. Frequently, patients had no school leaving certificate, and 30% of the patients had never started or never completed an apprenticeship. Getting along with galactosemia was rated as 'very good' or 'good' although following the diet was a burden. Social well-being and social functioning was lower compared to patients with PKU. DISCUSSION: Patients with galactosemia need a multi-professional team not only focusing on physical and/or biochemical aspects of disease but including also psycho-social dimensions of life.

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Year:  2012        PMID: 22447152     DOI: 10.1007/s10545-012-9469-y

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  20 in total

1.  High tolerance for oral galactose in classical galactosaemia: dietary implications.

Authors:  A M Bosch; H D Bakker; L J M de B Wenniger-Prick; R J A Wanders; F A Wijburg
Journal:  Arch Dis Child       Date:  2004-11       Impact factor: 3.791

2.  The adult galactosemic phenotype.

Authors:  Susan E Waisbren; Nancy L Potter; Catherine M Gordon; Robert C Green; Patricia Greenstein; Cynthia S Gubbels; Estela Rubio-Gozalbo; Donald Schomer; Corrine Welt; Vera Anastasoaie; Kali D'Anna; Jennifer Gentile; Chao-Yu Guo; Leah Hecht; Roberta Jackson; Bernadette M Jansma; Yijun Li; Va Lip; David T Miller; Michael Murray; Leslie Power; Nicolle Quinn; Frances Rohr; Yiping Shen; Amy Skinder-Meredith; Inge Timmers; Rachel Tunick; Ann Wessel; Bai-Lin Wu; Harvey Levy; Louis Elsas; Gerard T Berry
Journal:  J Inherit Metab Dis       Date:  2011-07-21       Impact factor: 4.982

3.  Intellectual development of the patients of the German Collaborative Study of children treated for phenylketonuria.

Authors:  P Burgard; E Schmidt; A Rupp; W Schneider; H J Bremer
Journal:  Eur J Pediatr       Date:  1996-07       Impact factor: 3.183

4.  Bone metabolism in galactosemia.

Authors:  B Panis; P Ph Forget; M J P G van Kroonenburgh; C Vermeer; P P Menheere; F H Nieman; M E Rubio-Gozalbo
Journal:  Bone       Date:  2004-10       Impact factor: 4.398

5.  Remarkable differences: the course of life of young adults with galactosaemia and PKU.

Authors:  A M Bosch; H Maurice-Stam; F A Wijburg; M A Grootenhuis
Journal:  J Inherit Metab Dis       Date:  2009-10-10       Impact factor: 4.982

6.  Bone mineral density in patients with classic galactosaemia.

Authors:  M E Rubio-Gozalbo; S Hamming; M J P G van Kroonenburgh; J A Bakker; C Vermeer; P Ph Forget
Journal:  Arch Dis Child       Date:  2002-07       Impact factor: 3.791

7.  Correlates of language impairment in children with galactosaemia.

Authors:  N L Potter; J-A C Lazarus; J M Johnson; R D Steiner; L D Shriberg
Journal:  J Inherit Metab Dis       Date:  2008-07-12       Impact factor: 4.982

8.  High phenylalanine levels directly affect mood and sustained attention in adults with phenylketonuria: a randomised, double-blind, placebo-controlled, crossover trial.

Authors:  Amber E ten Hoedt; Leo M J de Sonneville; Baudouin Francois; Nienke M ter Horst; Mirian C H Janssen; M Estela Rubio-Gozalbo; Frits A Wijburg; Carla E M Hollak; Annet M Bosch
Journal:  J Inherit Metab Dis       Date:  2010-12-10       Impact factor: 4.982

9.  Psychosocial developmental milestones in men with classic galactosemia.

Authors:  Cynthia Sophia Gubbels; Heleen Maurice-Stam; Gerard Thomas Berry; Annet Maria Bosch; Susan Waisbren; Maria Estela Rubio-Gozalbo; Martha Alexandra Grootenhuis
Journal:  J Inherit Metab Dis       Date:  2011-02-25       Impact factor: 4.982

10.  Evaluation of quality of life and description of the sociodemographic state in adolescent and young adult patients with phenylketonuria (PKU).

Authors:  Eva Simon; Martin Schwarz; Judith Roos; Nico Dragano; Max Geraedts; Johannes Siegrist; Gudrun Kamp; Udo Wendel
Journal:  Health Qual Life Outcomes       Date:  2008-03-26       Impact factor: 3.186

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  9 in total

Review 1.  Appropriateness of newborn screening for classic galactosaemia: a systematic review.

Authors:  L Varela-Lema; L Paz-Valinas; G Atienza-Merino; R Zubizarreta-Alberdi; R Vizoso Villares; M López-García
Journal:  J Inherit Metab Dis       Date:  2016-04-26       Impact factor: 4.982

2.  Novel mRNA-Based Therapy Reduces Toxic Galactose Metabolites and Overcomes Galactose Sensitivity in a Mouse Model of Classic Galactosemia.

Authors:  Bijina Balakrishnan; Ding An; Vi Nguyen; Christine DeAntonis; Paolo G V Martini; Kent Lai
Journal:  Mol Ther       Date:  2019-09-19       Impact factor: 11.454

3.  Management of adult patients with phenylketonuria: survey results from 24 countries.

Authors:  Friedrich K Trefz; Francjan J van Spronsen; Anita MacDonald; François Feillet; Ania C Muntau; Amaya Belanger-Quintana; Alberto Burlina; Mübeccel Demirkol; Marcello Giovannini; Christoph Gasteyger
Journal:  Eur J Pediatr       Date:  2014-12-06       Impact factor: 3.183

4.  Exploration of the Brain in Rest: Resting-State Functional MRI Abnormalities in Patients with Classic Galactosemia.

Authors:  Britt van Erven; Bernadette M Jansma; M Estela Rubio-Gozalbo; Inge Timmers
Journal:  Sci Rep       Date:  2017-08-22       Impact factor: 4.379

Review 5.  Sweet and sour: an update on classic galactosemia.

Authors:  Ana I Coelho; M Estela Rubio-Gozalbo; João B Vicente; Isabel Rivera
Journal:  J Inherit Metab Dis       Date:  2017-03-09       Impact factor: 4.982

6.  Classical galactosemia: neuropsychological and psychosocial functioning beyond intellectual abilities.

Authors:  Mendy M Welsink-Karssies; Kim J Oostrom; Merel E Hermans; Carla E M Hollak; Mirian C H Janssen; Janneke G Langendonk; Esmee Oussoren; M Estela Rubio Gozalbo; Maaike de Vries; Gert J Geurtsen; Annet M Bosch
Journal:  Orphanet J Rare Dis       Date:  2020-02-07       Impact factor: 4.123

7.  Qualitative interviews with adults with Classic Galactosemia and their caregivers: disease burden and challenges with daily living.

Authors:  Jason A Randall; Carolyn Sutter; Stella Wang; Evan Bailey; Lydia Raither; Riccardo Perfetti; Shoshana Shendelman; Claire Burbridge
Journal:  Orphanet J Rare Dis       Date:  2022-03-28       Impact factor: 4.123

Review 8.  Patient and observer reported outcome measures to evaluate health-related quality of life in inherited metabolic diseases: a scoping review.

Authors:  Carlota Pascoal; Sandra Brasil; Rita Francisco; Dorinda Marques-da-Silva; Agnes Rafalko; Jaak Jaeken; Paula A Videira; Luísa Barros; Vanessa Dos Reis Ferreira
Journal:  Orphanet J Rare Dis       Date:  2018-11-28       Impact factor: 4.123

Review 9.  Pathophysiology and management of classic galactosemic primary ovarian insufficiency.

Authors:  Synneva Hagen-Lillevik; John S Rushing; Leslie Appiah; Nicola Longo; Ashley Andrews; Kent Lai; Joshua Johnson
Journal:  Reprod Fertil       Date:  2021-06-25
  9 in total

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