The ASsessment and Physiotherapy managEment of Children with ataxia following surgical resection of posterior fossa Tumour (ASPECT study)

Abstract

Abstract
Helen Hartley, Thesis submitted for PhD (FOHSC), ASPECT Study
Background
Up to 70% of children present with ataxia (balance and co-ordination problems) following surgical resection of a posterior fossa tumour (PFT). Physiotherapy is a conventional management approach,
but few clinical research studies exist in this field to guide assessment and management.
Aims
1) Identify appropriate outcome measures to evaluate ataxia severity in children with PFT,
2) understand the current scope of physiotherapy practice and challenges to rehabilitation, and
3) determine the feasibility of conducting a randomised controlled trial (RCT) studying the effectiveness of virtual reality training (VRT) for balance in this population group.
Research Design
A mixed methods design consisting of four phases. Patient and Public Involvement was integral to all phases. Ethics approval was obtained.
Methods
Phase 1; Longitudinal observational cohort study examining the responsiveness of the Scale for the Assessment and Rating of Ataxia (SARA) and Brief Ataxia Rating Scale (BARS) and describing the natural history of ataxia in children with PFT.
Phase 2; Development and implementation of an e-survey to identify current international physiotherapy practice and challenges to rehabilitation.
Phase 3; Stakeholder workshops focused on the use of off-the-shelf VRT in children with PFT.
Phase 4; Mixed-methods (with embedded qualitative component) feasibility randomised controlled
trial examining usual care versus VRT and usual care in children with PFT.
Results
Phase 1; Both the SARA and BARS appeared to reflect change in ataxia, with the SARA scale potentially more sensitive to subtle change. Children with medulloblastoma and midline tumours presented with more balance and coordination problems.
Phase 2; Ninety-six physiotherapists (12 countries) completed the e-survey. Treatment approaches varied but consensus relating to treatment intensity was identified. Challenges to rehabilitation
included child and family factors, condition specific factors and service delivery factors.
Phase 3; Fifteen participants (children, their parents, innovators, physiotherapists) generated findings on engagement, practicality and utility of VRT for children with PFT and enabled game choice for Phase 4.
Phase 4; Ten children (aged 4-14 years), 1-3 years following surgery for PFT and nine parents participated. Recruitment was low (6%-40%, site dependent) but no issues with willingness for randomisation was identified. Assessment completion was high. Overall, the results suggest a future
definitive trial may be feasible; however, adaptation to recruitment strategies are likely to be necessary.
Conclusion
Key findings include the responsiveness of the SARA, risk factors for severe ataxia and the identification of multifactorial challenges to rehabilitation. Initial information regarding the feasibility
of conducting a formal randomised controlled trial examining VRT in children with PFT has been demonstrated, highlighting that further research is needed on trial protocol (including optimum timing
and dosage of intervention) and core sets of outcome measures.
Original Contributions to Knowledge
The key original contributions to knowledge within this field are: (1) Identification of gaps in the evidence base; (2) advance knowledge of sensitivity of outcome measures; (3) mapping current
physiotherapy practice; and (4) informing the design of future RCTs using VRT in children with PFT.

Date of Award	3 Feb 2022
Original language	English
Awarding Institution	Edge Hill University
Sponsors	National Institute for Health Research
Supervisor	BERNIE CARTER (Director of Studies), Ram Kumar (Supervisor), Lisa Bunn (Supervisor) & Elizabeth Cassidy (Supervisor)