• Maria M Wertli, Florian Brunner, Johann Steurer, and Ulrike Held.
• Horten Centre for Patient Oriented Research and Knowledge Transfer, University of Zurich, Pestalozzistrasse 24, Zurich, Switzerland.
• Plos One. 2017 Jan 1; 12 (3): e0173688.

BackgroundSince 2007, the Budapest criteria are recommended for the diagnosis of Complex Regional Pain Syndrome (CRPS) 1. The usefulness of bone scintigraphy (BS, index test) for the diagnosis of CRPS 1 remains controversial. Imperfect reference tests (RT) result in underestimation of the diagnostic accuracy of BS. Further, biased results can occur when a dependency between the RT and BS exists. The objective was to assess the impact of different RTs, specifically the Budapest criteria, and the assumed imperfect nature of the RT on the diagnostic accuracy of BS. Further, we analyzed the association between baseline characteristics and positive BS in patients with CRPS 1.MethodsSystematic literature review and Bayesian meta-analysis to assess the test accuracy of BS with and without accounting for the imperfect nature of the RT. We examined correlations (Spearman correlation coefficients / Wilcoxon tests) between baseline characteristics and the proportion of positive BS in patients with CRPS 1.ResultsThe pooled sensitivity was 0.804 (95% credible interval (CI) 0.225-1.0, 21 studies) and specificity 0.853 (95%CI 0.278-1.00). Sensitivity and specificity of BS increased when accounting for the imperfect nature of the RT. However, in studies using Budapest criteria as reference, the sensitivity decreased (0.551; 95% CI 0.046-1) and the specificity increased (0.935; 95% CI 0.306-1). Shorter disease duration and a higher proportion of males were associated with a higher proportion of positive BS (27 studies, disease duration <52 weeks Wilcoxon test p = 0.047, female proportion Spearman correlation -0.63, p = 0.009).ConclusionCompared to the accepted Budapest diagnostic criteria BS cannot be used to rule-in the diagnosis of CRPS 1. In patients with negative BS CRPS 1 is less likely the underlying illness. Studies using older or no diagnostic criteria should not be used to evaluate the diagnostic accuracy of BS in CRPS 1.

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