Abstract

ObjectiveTo characterize the high-resolution computed tomography (HRCT) pulmonary abnormalities in patients with synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome.MethodsPulmonary HRCT images were reconstructed from whole-spine computed tomography (CT) images of 67 patients with SAPHO syndrome. HRCT images of 58 healthy controls were also obtained and reviewed. Patients with pneumonia and tuberculosis were excluded. Demographic and clinical data such as gender, age, onset age, disease duration, erythrocyte sedimentation rate (ESR), highly sensitive C-reactive protein (hs-CRP) and the Bath Ankylosing Spondylitis Disease Activity Index (BASDAI) were collected from the SAPHO syndrome patients. Demographic characteristics, ESR and hs-CRP data from the healthy controls were also recorded. Student’s t test, Mann-Whitney U test, chi-squared test and logistic regression were employed to compare the HRCT findings of the two groups of patients.ResultsThe median age of the SAPHO syndrome patients was 47.0 years, interquartile range [38.0–53.0]; that of the healthy controls was 37.0[30.8–53.8]. From the detailed HRCT evaluations, abnormalities were identified in 45 patients. We found irregular linear opacities in 29 (43.3%) patients, opacities in 22 (32.8%), ground-glass opacity in 11 (16.4%), pleural thickening in 9 (13.4%), solitary nodules in 6 (9%), bronchiectasis in 3 (4.5%), pulmonary bulla in 2 (3%), multiple nodules in 1 (1.5%), and reticular patterns in 1 (1.5%). Compared to the healthy controls, the SAPHO syndrome patients had a significantly higher rate of opacities but a significantly lower percentage of nodules (especially multiple nodules), although the overall rates of abnormal HRCT findings were similar in the two groups. According to the multivariate logistic regression analysis, increased age and BASDAI < 4 were significant predictors of abnormal HRCT findings.ConclusionOur study is the first to address HRCT pulmonary abnormalities in SAPHO syndrome patients. SAPHO syndrome patients have a significantly higher percentage of opacities and a significantly lower rate of pulmonary nodules than healthy controls. BASDAI and age are possible good predictors of abnormal HRCT pulmonary findings.

Highlights

  • Synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome is a spectrum of heterogeneous diseases, including osteoarticular and dermatological manifestations, first proposed by Chamot et al in 1987[1]

  • From the detailed high-resolution computed tomography (HRCT) evaluations, abnormalities were identified in 45 patients

  • The overall rates of abnormal HRCT findings in the SAPHO syndrome patients and the reference group were similar, the distribution of abnormal findings differed between the two groups

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Summary

Introduction

Acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome is a spectrum of heterogeneous diseases, including osteoarticular and dermatological manifestations, first proposed by Chamot et al in 1987[1]. The core physiological changes that occur in SAPHO syndrome are osteitis and bone hypertrophy, which mainly manifest as pain and activity limitations in the affected area[2,3,4]. The major dermatological manifestations include palmoplantar pustulosis (PPP) and severe acne (SA), with psoriasis vulgaris considered a concomitant condition[5]. The pathogenesis of SAPHO syndrome has not yet been elucidated, and the means of early diagnosis and targeted therapy are still lacking. The prevalence of SAPHO syndrome was reported as 1/ 10,000 in Caucasians and 0.00144/100,000 in the Japanese population[6]

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