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Outcome measures for assessing change over time in studies of symptomatic children with hypermobility: a systematic
Muhammad Maarj1,2, Andrea Coda3,4, Louise Tofts5,6
1Narrabeen Sports Medicine Centre, Sydney Academy of Sport, Sydney, Australia. muhammad.maarj@uon.edu.au.
Insights
Generalized joint hypermobility (GJH) in children often causes symptoms. This review found no standard outcome measures for GJH lower limb symptoms, recommending standardized tools for pain, function, quality of life, and fatigue.
Area of Science:
- Pediatrics
- Rheumatology
- Clinical Trials
Background:
- Generalized joint hypermobility (GJH) is common in children.
- Approximately 20% of children with GJH experience associated symptoms.
- Lower limb symptoms are a frequent concern in pediatric GJH.
Purpose of the Study:
- To synthesize outcome measures used in studies of children with GJH and lower limb symptoms.
- To identify gaps in outcome measure standardization for pediatric GJH research.
- To provide recommendations for future research and clinical practice.
Main Methods:
- Systematic electronic database searches (Medline, CINAHL, Embase) up to March 2020.
- Inclusion of interventional and prospective longitudinal studies of children aged 5-18 years.
- Assessment of methodological quality using the Downs and Black checklist.
Main Results:
- Six studies (388 children) met the inclusion criteria.
- Twenty different instruments were used to measure pain, function, and quality of life.
- No specific outcome measures were validated for children with GJH and symptoms; fatigue was also assessed.
Conclusions:
- There is a lack of consensus on outcome measures for pediatric GJH with lower limb symptoms.
- Standardization of assessment tools in pediatric clinical trials is crucial.
- Recommended constructs for measurement include pain, function, quality of life, and fatigue, using validated, objective tools.
Background:
Generalised joint hypermobility (GJH) is highly prevalent among children and associated with symptoms in a fifth with the condition. This study aimed to synthesise outcome measures in interventional or prospective longitudinal studies of children with GJH and associated lower limb symptoms.
Methods:
Electronic searches of Medline, CINAHL and Embase databases from inception to 16th March 2020 were performed for studies of children with GJH and symptoms between 5 and 18 years reporting repeated outcome measures collected at least 4 weeks apart. Methodological quality of eligible studies were described using the Downs and Black checklist.
Results:
Six studies comprising of five interventional, and one prospective observational study (total of 388 children) met the inclusion criteria. Interventional study durations were between 2 and 3 months, with up to 10 months post-intervention follow-up, while the observational study spanned 3 years. Three main constructs of pain, function and quality of life were reported as primary outcome measures using 20 different instruments. All but one measure was validated in paediatric populations, but not specifically for children with GJH and symptoms. One study assessed fatigue, reporting disabling fatigue to be associated with higher pain intensity.
Conclusions:
There were no agreed sets of outcome measures used for children with GJH and symptoms. The standardisation of assessment tools across paediatric clinical trials is needed. Four constructs of pain, function, quality of life and fatigue are recommended to be included with agreed upon, validated, objective tools.
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