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Orthopaedic management of children with spinal dysraphism
Deepika Pinto1, Sabba Hussain2, Donato G Leo2,3
1Swansea Bay University Health Board, Swansea, UK.
Insights
Orthopaedic interventions for children with spinal dysraphism lack standardized outcome measures, hindering research consistency. Developing a core outcome set is crucial for reliable reporting in pediatric spinal dysraphism research.
Area of Science:
- Pediatric Orthopaedics
- Rehabilitation Medicine
- Clinical Trial Methodology
Background:
- Spinal dysraphism in children often leads to musculoskeletal deformities requiring orthopaedic interventions.
- These interventions are associated with significant patient morbidity and resource utilization.
- A need exists to standardize outcome reporting in pediatric spinal dysraphism care.
Conclusions:
- Reported outcomes for pediatric spinal dysraphism interventions are heterogeneous and often lack validation.
- The current reporting landscape limits research consistency and fails to capture patient perspectives.
- Development of a core outcome set is recommended to standardize reporting and advance research in this field.
Aims:
Children with spinal dysraphism can develop various musculoskeletal deformities, necessitating a range of orthopaedic interventions, causing significant morbidity, and making considerable demands on resources. This systematic review aimed to identify what outcome measures have been reported in the literature for children with spinal dysraphism who undergo orthopaedic interventions involving the lower limbs.
Methods:
A PROSPERO-registered systematic literature review was performed following PRISMA guidelines. All relevant studies published until January 2023 were identified. Individual outcomes and outcome measurement tools were extracted verbatim. The measurement tools were assessed for reliability and validity, and all outcomes were grouped according to the Outcome Measures Recommended for use in Randomized Clinical Trials (OMERACT) filters.
Results:
From 91 eligible studies, 27 individual outcomes were identified, including those related to clinical assessment (n = 12), mobility (n = 4), adverse events (n = 6), investigations (n = 4), and miscellaneous (n = 1). Ten outcome measurement tools were identified, of which Hoffer's Functional Ambulation Scale was the most commonly used. Several studies used unvalidated measurement tools originally developed for other conditions, and 26 studies developed new measurement tools. On the OMERACT filter, most outcomes reported pathophysiology and/or the impact on life. There were only six patient- or parent-reported outcomes, and none assessed the quality of life.
Conclusion:
The outcomes that were reported were heterogenous, lack validation and failed to incorporate patient or family perceptions. Until outcomes can be reported unequivocally, research in this area will remain limited. Our findings should guide the development of a core outcome set, which will allow consistency in the reporting of outcomes for this condition.
