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

Jie Lu,Xuewei Zhang,Chen Li,Wenrui Xu,Yanping Duan,Weihong Zhang,Hanzhang Lu,Rong Xue,Zhentao Zuo

doi:10.1186/s13023-017-0658-5

Abstract

BackgroundSynovitis-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.Results46.2% (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.ConclusionThese 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.

Highlights

Synovitis-acne-pustulosis-hyperostosis-osteitis (SAPHO) syndrome is a rare disease and there is no related literature concerning psychiatric symptoms in SAPHO patients
Clinical and Demographic characteristics All of the patients had typical characteristics of syndrome, such as osteoarticular and dermatological lesions, one example shown in Fig. 1, detailed data shown in Additional file 1: Table S1. 46.4% (13/28) of SAPHO patients were diagnosed with depression
Altered amplitude of low-frequency fluctuation (ALFF) in SAPHO patients Significant differences of the ALFF were revealed by analysis of variance (ANOVA) among the depressed SAPHO (D-SAPHO) patients, ND-SAPHO patients and normal controls (NC) in the following regions: bilateral frontal cortex, anterior cingulate cortex, temporal cortex and left inferior partial gyrus (Fig. 2)

Summary

Introduction

Synovitis-acne-pustulosis-hyperostosis-osteitis (SAPHO) syndrome is a rare disease and there is no related literature concerning psychiatric symptoms in SAPHO patients. 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). The ALFF reflects the extent of spontaneous neuronal activity [4], while the FC reveals the tendency of cortical networks to be co-activated [5] Both methods have been applied effectively to detect the mechanisms of pathophysiology of major depressive disorder (MDD)[6,7,8,9,10,11,12] and other mental disorders, such as autism spectrum disorders[13], schizophrenia[14], obsessive-compulsive disorders[15] and so on. A great variety of abnormal regions have been revealed in MDD, mainly including the prefrontal cortex, anterior cingulate cortex, cerebellum, amygdala and so on [17, 18]

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Discussion

Conclusion