Literature DB >> 27243819

How can clinical ethics guide the management of comorbidities in the child with Rett syndrome?

Jenny Downs1,2, David Forbes3, Michael Johnson4, Helen Leonard1.   

Abstract

Rett syndrome is a rare disorder caused by a mutation in the MECP2 gene. Those affected generally have severe functional impairments, and medical comorbidities such as scoliosis and poor growth are common. There is a paucity of information on the natural history of many rare disorders and an even greater deficit of evidence to guide best practice. The population-based and longitudinal Australian Rett Syndrome Database established in 1993 has supported investigations of the natural history of Rett syndrome and effectiveness of treatments. This paper reviews the disorder Rett syndrome and evidence for the management of scoliosis and poor growth within a clinical ethics framework. Compared with conservative management, we have shown that spinal fusion is associated with reduced mortality and better respiratory health. We have also shown that gastrostomy insertion is associated with subsequent weight gain. Family counselling for both procedures necessarily must include family perspectives and careful clinical attention to their needs and wishes. Vignettes describing family decision-making and experiences are presented to illustrate the principals of beneficence and autonomy in determining the best interests of the child and family. A blend of evidence-based practice with a strong clinical ethics framework has capacity to build existing strengths in families and reduce the negative impacts of disability and in so doing, optimise the health and wellbeing of those with Rett syndrome.
© 2016 Paediatrics and Child Health Division (The Royal Australasian College of Physicians).

Entities:  

Keywords:  Rett syndrome; clinical ethics; gastrostomy; rare disease; scoliosis

Mesh:

Year:  2016        PMID: 27243819      PMCID: PMC4990489          DOI: 10.1111/jpc.13241

Source DB:  PubMed          Journal:  J Paediatr Child Health        ISSN: 1034-4810            Impact factor:   1.954


  40 in total

1.  Spinal fusion in girls with Rett syndrome: post-operative recovery and family experiences.

Authors:  C Marr; H Leonard; I Torode; J Downs
Journal:  Child Care Health Dev       Date:  2015-03-09       Impact factor: 2.508

2.  Predictors of scoliosis in Rett syndrome.

Authors:  Sarah Ager; Susan Fyfe; John Christodoulou; Peter Jacoby; Lincoln Schmitt; Helen Leonard
Journal:  J Child Neurol       Date:  2006-09       Impact factor: 1.987

3.  Early decision of gastrostomy tube insertion in children with severe developmental disability: a current dilemma.

Authors:  C Martínez-Costa; S Borraz; C Benlloch; A López-Sáiz; V Sanchiz; J Brines
Journal:  J Hum Nutr Diet       Date:  2011-02-21       Impact factor: 3.089

4.  Guidelines for management of scoliosis in Rett syndrome patients based on expert consensus and clinical evidence.

Authors:  Jenny Downs; Anke Bergman; Philippa Carter; Alison Anderson; Greta M Palmer; David Roye; Harold van Bosse; Ami Bebbington; Eva Lena Larsson; Brian G Smith; Gordon Baikie; Sue Fyfe; Helen Leonard
Journal:  Spine (Phila Pa 1976)       Date:  2009-08-01       Impact factor: 3.468

5.  Gastrointestinal and nutritional problems occur frequently throughout life in girls and women with Rett syndrome.

Authors:  Kathleen J Motil; Erwin Caeg; Judy O Barrish; Suzanne Geerts; Jane B Lane; Alan K Percy; Fran Annese; Lauren McNair; Steven A Skinner; Hye-Seung Lee; Jeffrey L Neul; Daniel G Glaze
Journal:  J Pediatr Gastroenterol Nutr       Date:  2012-09       Impact factor: 2.839

6.  Early determinants of fractures in Rett syndrome.

Authors:  Jennepher Downs; Ami Bebbington; Helen Woodhead; Peter Jacoby; Le Jian; Amanda Jefferson; Helen Leonard
Journal:  Pediatrics       Date:  2008-03       Impact factor: 7.124

7.  Assessment and management of nutrition and growth in Rett syndrome.

Authors:  Helen Leonard; Madhur Ravikumara; Gordon Baikie; Nusrat Naseem; Carolyn Ellaway; Alan Percy; Suzanne Abraham; Suzanne Geerts; Jane Lane; Mary Jones; Katherine Bathgate; Jenny Downs
Journal:  J Pediatr Gastroenterol Nutr       Date:  2013-10       Impact factor: 2.839

Review 8.  Ethics in neurodevelopmental disability.

Authors:  Eric Racine; Emily Bell; Michael Shevell
Journal:  Handb Clin Neurol       Date:  2013

9.  Experience of gastrostomy using a quality care framework: the example of rett syndrome.

Authors:  Jenny Downs; Kingsley Wong; Madhur Ravikumara; Carolyn Ellaway; Elizabeth J Elliott; John Christodoulou; Peter Jacoby; Helen Leonard
Journal:  Medicine (Baltimore)       Date:  2014-12       Impact factor: 1.889

10.  Twenty years of surveillance in Rett syndrome: what does this tell us?

Authors:  Alison Anderson; Kingsley Wong; Peter Jacoby; Jenny Downs; Helen Leonard
Journal:  Orphanet J Rare Dis       Date:  2014-06-19       Impact factor: 4.123

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  1 in total

Review 1.  Clinical and biological progress over 50 years in Rett syndrome.

Authors:  Helen Leonard; Stuart Cobb; Jenny Downs
Journal:  Nat Rev Neurol       Date:  2016-12-09       Impact factor: 42.937

  1 in total

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