Inter-examiner reproducibility of tests and criteria for generalized joint hypermobility and benign joint hypermobility syndrome

Abstract
Objective. To test the reproducibility of tests and criteria for generalized joint hypermobility (GJH) and benign joint hypermobility syndrome (BJHS). Methods. A standardized protocol for clinical reproducibility studies was followed using a three-phase study (with a training, an overall agreement and a test phase). An overall agreement of at least 0.80 was required to proceed to the test phase. Phases 1, 2 and 3 used 14 patients (with varying degrees of hypermobility), 20 patients (50% cases) and 40 patients (50% cases), respectively. The inclusion criterion for cases was hypermobility (patients with Ehlers-Danlos Syndrome or BJHS) and for controls, non-hypermobility (patients with shoulder and/or back pain); patients were selected from patients’ files (phases 1 and 2) or included consecutively from our outpatient clinic (phase 3). Results. The overall agreement in phase 2 was 0.95 for GJH and 0.90 for BJHS. Reproducibility for diagnosing GJH and BJHS in phase 3 showed kappa values of 0.74 and 0.84, respectively. Kappa in the Beighton tests for diagnosing GJH (currently or historically) was generally above 0.80, except for the fifth fingers and elbows (≥0.60). In the Brighton tests for diagnosing BJHS, kappa was above 0.73, except for the skin signs (0.63). Lowest kappa was found in the Rotès-Quérol tests, where it was ≥0.57, except for the right shoulder (0.31). Conclusion. We found a good-to-excellent reproducibility of tests and criteria for GJH and BJHS. Future research on the validity of the tests and criteria for joint hypermobility is urgently needed.