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Orphanet Journal of Rare Diseases

, 12:103

First Online: 25 May 2017Received: 16 March 2017Accepted: 18 May 2017DOI: 10.1186-s13023-017-0658-5

Cite this article as: Lu, J., Duan, Y., Zuo, Z. et al. Orphanet J Rare Dis 2017 12: 103. doi:10.1186-s13023-017-0658-5


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.

KeywordsSAPHO syndrome Depression Resting state functional magnetic resonance imaging rs-fMRI Default mode network DMN AbbreviationsSAPHOSynovitis-acne-pustulosis-hyperostosis-osteitis

rs-fMRIResting state functional magnetic resonance imaging

BOLDBlood oxygenation level dependent

ALFFAmplitude of low-frequency fluctuation

FCFunctional connectivity

MDDMajor depressive episode

DMNThe default mode network


ASAnkylosing spondylitis

PsAPsoriatic arthritis

VASVisual Analogue Scale

BASDAIBath Ankylosing Spondylitis Disease Activity Index

BASFIBath Ankylosing Spondylitis Functional Index

NSAIDsNonsteroidal anti-inflammatory drugs

DMARDsDisease-modifying antirheumatic drugs

NCNormal controls

M.I.N.IThe Mini-International Neuropsychiatric Interview

HDRSThe 17-item Hamilton Depression Rating Scale


ND-SAPHONon-depressed SAPHO

TRRepetition time

TEEcho time

FOVField of view

DPARSFData Processing Assistant for Resting-State fMRI

RESTResting-State fMRI Data Analysis Toolkit 1.8

ANCOVAAnalysis of covariance

VLPFCVentrolateral prefrontal cortex

DLPFCDorsolateral prefrontal cortex

GMDGray matter density

ReHoRegional homogeneity

VMPFCVentromedial prefrontal cortex

Electronic supplementary materialThe online version of this article doi:10.1186-s13023-017-0658-5 contains supplementary material, which is available to authorized users.

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


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