Depression in patients with SAPHO syndrome and its relationship with brain activity and connectivity

BACKGROUND: Synovitis-acne-pustulosis-hyperostosis-osteitis (SAPHO) syndrome is a rare disease and there is no related literature concerning psychiatric symptoms in SAPHO patients. Thus, we believe that this will be the first paper to explore the episode and the neurobiological basis of depression symptoms in SAPHO patients using resting state functional magnetic resonance imaging (rs-fMRI). Twenty-eight SAPHO patients and fifteen age- and gender- matched normal controls (NC) were consecutively submitted to psychiatric evaluation and rs-fMRI scanning. RESULTS: 46.2% (13/28) of SAPHO patients were diagnosed as depression. The local spontaneous activity study showed that depressed SAPHO (D-SAPHO) patients had decreased amplitude of low-frequency fluctuation (ALFF) in the bilateral ventrolateral prefrontal cortex (VLPFC, attributed to the anatomical structures of Brodmann’s area 47, 45 and 44) and right dorsolateral prefrontal cortex (DLPFC, attributed to the anatomical structures of Brodmann’s area 8, 9 and 46), increased ALFF in the bilateral middle temporal gyrus, when compared to non-depressed SAPHO (ND-SAPHO) patients. The functional connectivity (FC) study disclosed that D-SAPHO patients had an increased FC in the anterior portions of default mode network (DMN) (the bilateral inferior frontal cortex, anterior cingulate cortex and insula cortex), and a decreased FC in the posterior areas of DMN (left middle occipital cortex), when compared to ND-SAPHO patients. Furthermore, correlation analysis revealed that both ALFF and FC values were significantly correlated with depression scores of SAPHO patients. CONCLUSION: These results prompt us to understand the underlying pathophysiological mechanism of depression in SAPHO syndrome, and demonstrate that abnormal brain functional areas may serve as effective biological indicators to monitor depression in the future. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (doi:10.1186/s13023-017-0658-5) contains supplementary material, which is available to authorized users.