Bone Fragility in Hereditary Connective Tissue Disorders: A Systematic Review and Meta-Analysis
Pooled result · Pooled prevalence of fractureResult — go to the resultPooled odds ratio 4.90 95% CI 1.49–16.08Authors' conclusion
EDS was associated with fracture, which could be independent of osteoporosis status. MFS had a milder degree of increased risk of fracture and osteoporosis. Despite no data from cohort studies, there was a significantly higher rate of fracture in LDS.
Study context
- Population
- Patients with hereditary connective tissue disorders (HCTD): Ehlers-Danlos syndrome (EDS), Marfan's syndrome (MFS) and Loeys-Dietz syndrome (LDS); Medline and EMBASE to June 202219 studies
- Comparison
- HCTD vs healthy individuals
- Outcome
- Pooled prevalence of fracture
Methodological details4206 articles identified; generic inverse variance method
- 4206 articles identified; generic inverse variance method
Pooled result
As reported in the abstract
Pooled odds ratio
4.90
95% CI 1.49–16.08
By group
EDSvs Healthy individuals
pooled OR 4.90 (95% CI 1.49–16.08)
Further pooled results
| Outcome | EDS | MFS | Estimate | |
|---|---|---|---|---|
| Pooled prevalence of osteoporosisEDSvs Healthy individualsI2 87% | 17% (95% CI 8%–34%) | pooled OR 1.34 (95% CI 0.28–6.36) | ||
| I2 | 96% | — not stated in the abstract | ||
| MFS: osteoporosis prevalence, fracture and osteoporosis risk (one study)One study | — not stated in the abstract | 5% (95% CI 3%–8%) | IRR (fracture) 1.35 (95% CI 1.18–1.55) | |
| MFS: osteoporosis prevalence, fracture and osteoporosis risk (one study)One study | subhazard ratio (osteoporosis) 3.97 (95% CI 2.53–6.25) | |||
| 0.10.31310Ratio (log scale) |
— not stated in the abstract
Original publication
Bone Fragility in Hereditary Connective Tissue Disorders: A Systematic Review and Meta-Analysis. Endocr Pract. Published 17 February 2023. doi:10. 1016/ j. eprac. 2023. 02. 003
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