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The clinical effectiveness and personal experience of supported standing for children with cerebral palsy: a comprehensive systematic review protocol

Source:

JBI Evidence Synthesis

July 2014, Volume :12 Number 7 , page 101 - 118 [Free]

Authors

  1. Freeman, Jennifer PhD
  2. Marsden, Jonathan PhD
  3. Rapson, Rachel MSc
  4. Kent, Bridie PhD

Abstract

Review question/objective: The overall aim of this systematic review is to explore the uptake, clinical effectiveness, and personal experience of standing frame use for children with CP.

 

The quantitative objectives are to identify:

 

the clinical effectiveness of standing frame use for children with CP on impairments, activities, participations, and quality of life.

 

the level of uptake of, and adherence to, regular standing frame programs

 

 

The qualitative objectives are to identify, for children with CP, and their families/caregivers:

 

* their experience of using a standing frame program

 

* the enablers and barriers of using a standing frame program

 

 

Background: Cerebral palsy is the most frequent cause of motor disability in children and adolescents1 with a prevalence of 2 to 3 children per 1000 live births. It is defined as "a group of permanent disorders of the development of movement and posture, causing activity limitations that are attributed to non-progressive disturbances that occurred in the developing fetal or infant brain. The motor disorders of CP are often accompanied by disturbances of sensation, perception, cognition, communication, and behavior by epilepsy, and by secondary musculoskeletal problems").2(p.9) Although the initial damage to the central nervous system does not progress, over time, as the child develops symptoms can progress. For this reason a definitive diagnosis of CP is often not provided initially and can vary from 18 months to five years depending on presentation.2

 

Cerebral Palsy affects the development of posture and movement. The severity of impairments is extremely varied. Motor symptoms include spasticity, cocontraction, loss of selective muscle control, and muscle weakness. If left unchecked they can lead to progressive musculoskeletal problems such as hip dysplasia, contracture and scoliosis.3 Based on evidence from large databases describing disease characteristics, it is estimated that approximately 35% of children are unable to stand independently, four percent are unable to stand at all, and almost one third of children are non-ambulant.4 These children spend most of their lives in a sitting or lying position and hence are at the particular risk of progression of these and additional deficits such as increased muscle stiffness, increased weakness, loss of range of movement and changes in bone development such as osteoporosis. Both primary deficits and their sequelae can have a significant impact on functional ability and CP is associated with an estimated [Euro sign]0.8 million in social and healthcare costs across the lifespan and increased carer burden.5,6

 

In the UK assisted standing programs are routinely employed as part of the postural management approach for children with cerebral palsy who are unable to independently maintain well aligned standing or who have limited mobility in the upright position.7 A standing program refers to the use of adaptive equipment, for example, a standing frame that provides external, adjustable support to facilitate an upright position. The aim of these standing programs is to prevent or minimize motor symptoms, to encourage proper alignment, and to improve bone growth and bone mineral density through mechanical loading of the lower limbs and spine.8 A range of clinical benefits have been demonstrated which include; improved acetabular formation, prevention of hip dysplasia and improvements in bone density and gross motor development.8-14 Anecdotally, physiotherapists also site a number of additional benefits of assisted standing including: pressure relief, improved joint range of motion, soft tissue flexibility, promoting self-esteem and social interaction as well as accessing educational materials.15

 

The introduction of postural management programs, such as supported standing, into the daily lives of children and their family unit requires commitment from both the child and their carer16, hence practical difficulties with implementing the activity may arise because of issues of acceptance, logistics, and tolerance.17 This is particularly pertinent when children are more severely disabled, the very group who are typically targeted for supported standing frame programs. Importantly, previous work indicates that the level of compliance in their involvement in supported standing programs may be a key factor in determining clinical outcomes.18 This highlights the importance of understanding the experiences of children, their family and carers, and gaining insight into the enablers and barriers of their compliance in participating in these programs in order to both inform service provision19 and the design of research studies aiming to evaluate their effectiveness.

 

Currently there is little guidance regarding standing children with CP so individual health professionals (usually physiotherapists) are forced to decide the frequency, duration, type of standing regimen, and frame based on their clinical experience. In response to this, the National Institute of Clinical Excellence20 has identified the need for further research to investigate the clinical effectiveness of standing programs, particularly in the younger child, and in relation to the different durations of standing programs.

 

Prior to undertaking further research, it is important to undertake a comprehensive review of the literature about the current state of evidence, hence the identified need for this systematic review (SR). An initial search of the literature identified two SRs examining studies which evaluated pediatric supported standing programs21,22; however, these reviews were not restricted to children with CP, instead including any population of children, and the search timeframes were restricted to trials published prior to October 2009 and August 2012 respectively. Also identified in our initial review was one SR specifically examining interventions targeted at children with CP and low bone mineral density10 which included three clinical trials of standing frame use in children with CP published prior to 2009. A further two SRs were unearthed which examined studies evaluating the effectiveness of common physical therapy interventions; however neither of these reviews included standing programs.23,24

 

When comparing the results of the initial literature search of quantitative studies with the reference lists of Paleg, Glickman and Hough's SRs,10, 21, 22 it was noted that there were studies which had evaluated standing as an intervention in children with CP that had not been included in any of these reviews, namely a randomized controlled trial (RCT)25 and an observational study14; their lack of inclusion reflecting the time restrictions for the search strategies utilized in these SRs.10, 22 In addition, our initial search revealed one registered clinical trial26, which has the potential to publish data within the time frame of this SR. This supports the need for an updated SR regarding the clinical effectiveness of standing frame use with children with CP from a quantitative perspective.

 

With regard to people's experience in relation to participating in the standing frame program, our initial search suggests that factors such as positioning, comfort and personal acceptance can significantly impact theduration and frequency of standing frame use.13, 16, 27These findings are similar to those expressed by parents of children with CP in qualitative research of closely related fields such as physiotherapy exercise programs.28 Furthermore, qualitative research in the related area of walking29 suggests that, as well as having physiological and functional benefits, standing may hold symbolic significance for both the child and their parents, which may also influence the level of uptake and compliance with standing frame programs. The literature search failed to identify any meta-synthesis of qualitative studies in this area, hence justifying the need for this to be incorporated within this proposed SR.

 

In conclusion, it is suggested that this proposed SR will provide novel information. It is specific to children and adolescents with CP, will include studies other than RCTs, will only cover primary studies (not reviews), and will include a meta-synthesis of qualitative studies.

 

Article Content

Quantitative component inclusion criteria

Types of participants

Quantitative studies that include children and adolescents (aged < 18 years) with CP will be considered.

 

Types of intervention(s)

Studies that evaluate standing frame interventions in comparison to either usual care or other specific physiotherapy interventions will be considered.

 

Types of outcomes

The International Classification of Functioning, Disability and Health (ICF) will be used as the framework on which to classify the outcomes measured in the reviewed studies. Many physiotherapy approaches or interventions are directed at the "body functions and structures" domain of the ICF30 with the aim of hopefully improving the activities and participation of the child. However, it is often not known whether improvements at the impairment level will transfer into functional gains or gains to a child's level of participation. This SR will attempt to identify whether this is the case. To ensure categorization to the correct ICF domain we will use the http://apps.who.int/classifications/icfbrowser/ tool.

 

Listed below are the outcomes of interest:-

 

Impairments of the child and adolescent with CP:

 

* contracture

 

* range of motion

 

* spasticity

 

* spasms

 

* muscle strength

 

* muscle bulk

 

* pain

 

* balance

 

* pressure areas/pressure sores

 

* hip migration index

 

* bone mineral density

 

* bladder symptoms

 

* bowel symptoms/bowel evacuation

 

* sensory

 

* respiration

 

* confidence

 

* self-esteem

 

 

Activities of the child and adolescent with CP

 

* motor function

 

* standing ability

 

* activities of daily living

 

* walking/gait

 

* standing

 

* transfers

 

* mobility

 

* communication

 

* upper limb function

 

* head control

 

 

Participations of the child and adolescent with CP

 

* community mobility

 

* education

 

* leisure pursuits

 

* social life

 

 

In addition, the following outcomes data will also be extracted:

 

* carer burden/caregiver distress

 

* quality of life

 

* adverse events, fracture incidence, respiratory complications, mortality of the child and adolescent with CP.

 

* standing compliance/adherence

 

 

Types of studies

Both experimental and epidemiological study designs including randomized controlled trials, non-randomized controlled trials, quasi-experimental, before and after studies, prospective and retrospective cohort studies, case control studies and analytical cross sectional studies will be considered in the review.

 

Descriptive epidemiological study designs including case series, individual case reports and descriptive cross sectional studies will also be considered.

 

Qualitative component inclusion criteria

Types of participants

This component will consider studies that include children and adolescents (aged < 18 years) with CP and/or their family members or other caregivers.

 

Phenomena of interest

Studies that investigate the experiences of families, children with CP, and other caregivers who are involved with postural management programs which include a standing frame component will be considered.

 

Context

Studies that explore the experiences of participants using a standing frame program within acute care, primary health care, or community settings will be considered.

 

Types of studies

Studies that focus on qualitative data including, but not limited to, designs such as phenomenology, grounded theory, ethnography, action research will be considered.

 

Search strategy

The search strategy aims to find both published and unpublished studies. A three-step search strategy will be utilized in this review. An initial limited search of MEDLINE, AMED, CINAHL and EMBASE will be undertaken followed by analysis of the text words contained in the title and abstract, and of the index terms used to describe articles. A second search using all identified keywords and index terms will then be undertaken across all included databases. Thirdly, the reference list of all identified reports and articles will be searched for additional studies. Studies published in English will be considered for inclusion in this review. There is no restriction of dates because it is unclear when the earliest research on this topic was available.

 

The databases to be searched include:

 

MEDLINE (OVID)

 

EMBASE (OVID)

 

The Cochrane Central Register of Controlled Trials

 

CINAHL

 

AMED (EBSCO)

 

PEDro

 

The search for unpublished studies will include scanning of references of identified studies, unpublished studies, and grey literature. It will be undertaken using: google scholar, and Conference Papers Index (http://www.refworks-cos.com/cpi/) and a search of clinical trials registers via http://www.controlled-trials.com and http://clinicaltrials.gov

 

Initial keywords to be used will be:

 

child or adolescent or (child*, adolex*) and

 

cerebral palsy or (hemiplegi*, diplegi*, quadriplegi*, monoplegi*, triplegi*)

 

standing frame or (stand*, standing, standing apparatus, passive standing)

 

postural management

 

weight bearing

 

tilt table

 

qualitative

 

quantitative

 

An example of the search strategy for Medline (OVID) is included below:

 

"cerebral palsy".mp. [mp=title, abstract, subject headings, heading word, drug trade name, original title, device manufacturer, drug manufacturer, device trade name, keyword]. limit to (human and english language)

 

"standing frame".mp. [mp=title, abstract, subject headings, heading word, drug trade name, original title, device manufacturer, drug manufacturer, device trade name, keyword]. limit to (human and english language)

 

"stand*".mp. [mp=title, abstract, subject headings, heading word, drug trade name, original title, device manufacturer, drug manufacturer, device trade name, keyword]. limit to (human and english language)

 

"standing apparatus".mp. [mp=title, abstract, subject headings, heading word, drug trade name, original title, device manufacturer, drug manufacturer, device trade name, keyword]. limit to (human and english language)

 

"passive standing".mp. [mp=title, abstract, subject headings, heading word, drug trade name, original title, device manufacturer, drug manufacturer, device trade name, keyword]. limit to (human and english language)

 

"postural management".mp. [mp=title, abstract, subject headings, heading word, drug trade name, original title, device manufacturer, drug manufacturer, device trade name, keyword]. limit to (human and english language)

 

"tilt table".mp. [mp=title, abstract, subject headings, heading word, drug trade name, original title, device manufacturer, drug manufacturer, device trade name, keyword]. limit to (human and english language)

 

"weight bearing".mp. [mp=title, abstract, subject headings, heading word, drug trade name, original title, device manufacturer, drug manufacturer, device trade name, keyword] limit to (human and english language)

 

1 AND (2 OR 3 OR 4 OR 5 OR 6 OR 7 OR 8)

 

 

Assessment of methodological quality

Quantitative papers selected for retrieval will be assessed by two independent reviewers for methodological validity prior to inclusion in the review using the standardized critical appraisal instrument, the Joanna Briggs Institute Meta-Analysis of Statistics Assessment and Review Instrument (JBI-MAStARI) (Appendix I). Any disagreements that arise between the reviewers will be resolved through discussion or with a third reviewer.

 

Qualitative papers selected for retrieval will be assessed by two independent reviewers for methodological validity prior to inclusion in the review using the standardized critical appraisal instrument, the Joanna Briggs Institute Qualitative Assessment and Review Instrument (JBI-QARI) (Appendix I). Any disagreements that arise between the reviewers will be resolved through discussion or with a third reviewer.

 

Data collection

Quantitative data will be extracted from papers included in the review using the standardized data extraction tool from JBI-MAStARI (Appendix II). The data, to be extracted independently by two people, will include specific details about the interventions, populations, study methods and outcomes of significance to the review question and specific objectives. Authors of primary studies will be contacted for missing information or to clarify data.

 

Qualitative data will be similarly extracted from papers included in the review by two people independently using the standardized data extraction tool from JBI-QARI (Appendix II). The data extracted will include specific details about the interventions, populations, study methods and outcomes of significance to the review question and specific objectives.

 

Data synthesis

Quantitative papers will, where possible, be pooled in statistical meta-analysis using JBI-MAStARI. All results will be subject to double data entry. Effect sizes, expressed as odds ratio (for categorical data) and weighted mean differences (for continuous data), and their 95% confidence intervals will be calculated for analysis. Heterogeneity will be assessed statistically using the standard Chi-square and also explored using subgroup analyses based on the different quantitative study designs included in this review. Where statistical pooling is not possible the findings will be presented in narrative form including tables and figures to aid in data presentation where appropriate.

 

Qualitative research findings will, where possible, be pooled using JBI-QARI. This will involve the aggregation or synthesis of findings to generate a set of statements that represent that aggregation, through assembling the findings (Level 1 findings) rated according to their quality, and categorizing these findings on the basis of similarity in meaning (Level 2 findings). These categories are then subjected to a meta-synthesis in order to produce a single comprehensive set of synthesized findings (Level 3 findings) that can be used as a basis for evidence-based practice. Where textual pooling is not possible the findings will be presented in narrative form.

 

Conflicts of interest

There are no conflicts of interest for any of the authors

 

References

 

1. Prevalence and characteristics of children with cerebral palsy in Europe. Developmental Medicine & Child Neurology. 2002;44(9):633-40. [Context Link]

 

2. Rosenbaum P, Paneth N, Leviton A, Goldstein M, Bax M, Damiano D, et al. A report: the definition and classification of cerebral palsy April 2006. Developmental Medicine and Child Neurology Supplement. 2007;109:8-14. [Context Link]

 

3. Morrell DS, Pearson JM, Sauser DD. Progressive bone and joint abnormalities of the spine and lower extremities in cerebral palsy. Radiographics: A Review Publication Of The Radiological Society Of North America, Inc. 2002;22(2):257-68. [Context Link]

 

4. Rodby-Bousquet E, Hagglund G. Sitting and standing performance in a total population of children with cerebral palsy: a cross-sectional study. BMC Musculoskelet Disord. 2010;11(131):1471-2474. [Context Link]

 

5. Kruse M, Michelsen SI, Flachs EM, Bronnum-Hansen H, Madsen M, Uldall P. Lifetime costs of cerebral palsy. Dev Med Child Neurol. 2009;51(8):622-8. [Context Link]

 

6. Majnemer A, Shevell M, Law M, Poulin C, Rosenbaum P. Indicators of distress in families of children with cerebral palsy. Disabil Rehabil. 2012;34(14):1202-7. [Context Link]

 

7. Gudjonsdottir B, Mercer VS. Effects of a dynamic versus a static prone stander on bone mineral density and behavior in four children with severe cerebral palsy. Pediatric Physical Therapy. 2002;14(1):38-46. [Context Link]

 

8. Salem Y, Lovelace-Chandler V, Zabel RJ, McMillan AG. Effects of prolonged standing on gait in children with spastic cerebral palsy. Physical and Occupational Therapy in Pediatrics. 2010;30(1):54-65. [Context Link]

 

9. Caulton JM, Ward KA, Alsop CW, Dunn G, Adams JE, Mughal MZ. A randomised controlled trial of standing programme on bone mineral density in non-ambulant children with cerebral palsy. Archives of Disease in Childhood. 2004;89(2):131-5. [Context Link]

 

10. Hough JP, Boyd RN, Keating JL. Systematic review of interventions for low bone mineral density in children with cerebral palsy. Pediatrics. 2010;125(3):e670-e8. [Context Link]

 

11. Pountney T, Mandy A, Green E, Gard P. Management of hip dislocation with postural management. Child Care Health Dev. 2002;28(2):179-85. [Context Link]

 

12. Pountney T, Green EM. Hip dislocation in cerebral palsy. BMJ. 2006;332(7544):772-5. [Context Link]

 

13. Gibson SK, Sprod JA, Maher CA. The use of standing frames for contracture management for nonmobile children with cerebral palsy. International Journal of Rehabilitation Research. 2009;32(4):316-23. [Context Link]

 

14. Damcott M, Blochlinger S, Foulds R. Effects of passive versus dynamic loading interventions on bone health in children who are nonambulatory. Pediatr Phys Ther. 2013;25(3):248-55. [Context Link]

 

15. Taylor K. Factors affecting prescription and implementation of standing-frame programs by school-based physical therapists for children with impaired mobility. Pediatr Phys Ther. 2009;21(3):282-8. [Context Link]

 

16. Bush S. Assisted standing-does children's experience match therapists goals? Developmental Medicine And Child Neurology. 2009;51:15-6. [Context Link]

 

17. Fowler EG, Kolobe TH, Damiano DL, Thorpe DE, Morgan DW, Brunstrom JE, et al. Promotion of physical fitness and prevention of secondary conditions for children with cerebral palsy: section on pediatrics research summit proceedings. Phy Ther. 2007;87(11):1495-510. [Context Link]

 

18. Pountney TE, Mandy A, Green E, Gard PR. Hip subluxation and dislocation in cerebral palsy - A prospective study on the effectiveness of postural management programmes. Physiotherapy Research International. 2009;14(2):116-27. [Context Link]

 

19. Kruijsen-Terpstra AJ, Ketelaar M, Boeije H, Jongmans MJ, Gorter JW, Verheijden J, et al. Parents' experiences with physical and occupational therapy for their young child with cerebral palsy: a mixed studies review. Child Care Health Dev. 2013. [Context Link]

 

20. Mugglestone MA, Eunson P, Murphy MS. Spasticity in children and young people with non-progressive brain disorders: summary of NICE guidance. BMJ. 2012;345:e4845. [Context Link]

 

21. Glickman LB, Geigle PR, Paleg GS. A systematic review of supported standing programs. J Pediatr Rehabil Med. 2010;3(3):197-213. [Context Link]

 

22. Paleg GS, Smith BA, Glickman LB. Systematic Review and Evidence-Based Clinical Recommendations for Dosing of Pediatric Supported Standing Programs. Pediatric Physical Therapy. 2013;25(3):232-47 10.1097/PEP.0b013e318299d5e7. [Context Link]

 

23. Anttila H, Autti-Ramo I, Suoranta J, Makela M, Malmivaara A. Effectiveness of physical therapy interventions for children with cerebral palsy: a systematic review. BMC Pediatr. 2008;8:14. [Context Link]

 

24. Martin L, Baker R, Harvey A. A systematic review of common physiotherapy interventions in school-aged children with cerebral palsy. Phys Occup Ther Pediatr. 2010;30(4):294-312. [Context Link]

 

25. Costi et al, Effectiveness of Standing Frame on Constipation: Clinical Trials.gov IdentifierNCT01811559. Estimated completion date Feb 2014. [Context Link]

 

26. Lee B-K, Chon S-C. Effect of whole body vibration training on mobility in children with cerebral palsy: a randomized controlled experimenter-blinded study. Clinical Rehabilitation. 2013;27(7):599-607. [Context Link]

 

27. Daniels N, Pinnington L, Clift M, Gopsill C, Armstrong J, Ward C. A review of standing frame features and their impact on user acceptability. International Journal of Therapy & Rehabilitation. 2005;12(8):363. [Context Link]

 

28. Peplow UC, Carpenter C. Perceptions of parents of children with cerebral palsy about the relevance of, and adherence to, exercise programs: a qualitative study. Phys Occup Ther Pediatr. 2013;33(3):285-99. [Context Link]

 

29. Gibson BE, Teachman G, Wright V, Fehlings D, Young NL, McKeever P. Children's and parents' beliefs regarding the value of walking: rehabilitation implications for children with cerebral palsy. Child: Care, Health and Development. 2012;38(1):61-9. [Context Link]

 

30. Jette AM. Toward a Common Language for Function, Disability, and Health. Phys Ther. 2006;86(5):726-34. [Context Link]

Appendix I: Appraisal instruments

 

QARI appraisal instrument

 

MAStARI appraisal instruments[Context Link]

Appendix II: Data extraction instruments

 

QARI data extraction instrument

 

MAStARI data extraction instrument[Context Link]

 

Keywords: standing; cerebral palsy; developmental delay; physiotherapy; postural management

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