Three patterns of osteoarticular involvement in SAPHO syndrome: a cluster analysis based on whole body bone scintigraphy of 157 patients

Cao, Yihan; Li, Chen; Yang, Qiao; Wu, Nan; Xu, Ping; Li, Yueting; Shi, Ximin; Pan, Qingqing; Wu, Xia; Sun, Xiaochuan; Zhao, Yanyang; Gu, Zhaodi; Yuan, Siyi; Zuo, Yuzhi; Li, Li; Zhang, Weihong; Hao, Weixin; Liu, Jinhe; Jing, Hongli; Zhang, Wen

doi:10.1093/rheumatology/key415

Cited by 29 publications

(27 citation statements)

References 17 publications

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“…In our study, the most frequently involved sites were the SClJ and the first rib area. Such findings were consistent with a study by Cao et al based on 99m Tc-MDP WBBS [5].…”

Section: Discussionsupporting

confidence: 94%

“…Diagnostic challenges may arise due to heterogeneous manifestations of the disease: either acne and arthritis or acne and anterior chest wall (ACW) osteitis with an unclear pustulosis history [3]. The ACW is the most commonly involved site, affecting approximately 70-90% of adult patients [4][5][6][7][8][9]. The radiological findings of the osteoarticular manifestations in SAPHO syndrome play a key role in its early and correct diagnosis [10].…”

Section: Introductionmentioning

confidence: 99%

“…The radiological findings of the osteoarticular manifestations in SAPHO syndrome play a key role in its early and correct diagnosis [10]. The typical "bull's head" change on whole-body bone scintigraphy (WBBS) is characteristic of SAPHO syndrome [4], but the frequency of the sign is low [5]. Conventional X-ray and computed tomography examinations have limited capacity in visualizing active inflammatory changes [4].…”

Section: Introductionmentioning

confidence: 99%

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Anterior chest wall in SAPHO syndrome: magnetic resonance imaging findings

Cao

et al. 2020

Arthritis Res Ther

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Background The anterior chest wall (ACW) involvement is characteristic of synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome, yet little research has focused on its magnetic resonance imaging (MRI) findings. Purpose To characterize the MRI features of the ACW in patients with SAPHO syndrome. Methods Seventy-one patients with SAPHO syndrome and ACW involvement evidenced by bone scintigraphy were recruited in this cross-sectional study. The ACW region was scanned using sagittal, axial, and oblique coronal Dixon T2-weighted sequences and axial Dixon T1-weighted sequences. The characteristics of both active inflammatory and chronic structural lesions were evaluated. Results The ACW lesions exhibited an asymmetrical distribution and a predilection for the sternocostoclavicular region (93.0%). Notably, 91.5% of the patients had lesions in the area of the anterior first ribs. Bone marrow edema (BME) was observed in 63 (88.7%) patients, which mainly affected the sternocostal joints (87.3%) and the manubrium sterni (84.5%). All of the BMEs were distributed under the articular surface or the bone cortex, consistent with the distribution of the ligaments and joint capsules. Synovitis was detected in 64 (90.1%) patients, with a predilection for the sternoclavicular joints (76.1%). A soft tissue mass or infiltration was found in all the patients who had bone marrow edema. Thirteen (18.3%) patients showed venous stenosis. Structural changes included bone bridge formation (80.3%), hyperostosis (43.7%), and fat infiltration (39.4%). Four common patterns of involvement were observed: the first rib area, the sternoclavicular area, the sternal angle area, and the areas of the second to sixth sternocostal joints. Conclusion The ACW lesions of SAPHO syndrome demonstrated a triad of enthesitis, synovitis, and osteitis, suggesting complex interactions among the ligaments, synovium, and bones in the region. The inflammatory changes in the first rib area were highlighted in SAPHO syndrome.

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“…In our study, the most frequently involved sites were the SClJ and the first rib area. Such findings were consistent with a study by Cao et al based on 99m Tc-MDP WBBS [5].…”

Section: Discussionsupporting

confidence: 94%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Anterior chest wall in SAPHO syndrome: magnetic resonance imaging findings

Cao

et al. 2020

Arthritis Res Ther

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“…59 Based on WBBS manifestations, the osteoarticular involvement in SAPHO syndrome can be categorized into three distinct patterns, that is, the spinal type, the costal type and the sternoclavicular type. 64 However, both active and chronic lesions manifest as high uptake areas on WBBS, which means WBBS cannot determine the disease activity of lesions.…”

Section: Imaging Assessmentmentioning

confidence: 99%

Synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: review and update

Liu

Tang

Cao

et al. 2020

Therapeutic Advances in Musculoskeletal

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Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a spectrum of heterogeneous diseases characterized by osteoarticular and dermatological manifestations. Osteitis and hyperostosis are core clinical manifestations in SAPHO syndrome, typically affecting multiple areas and possibly progressing to irreversible osteoarticular damage. Most patients with SAPHO have cutaneous involvement, mainly manifested as palmoplantar pustulosis and severe acne. Systemic manifestations are uncommon but occasionally reported. Epidemiological studies suggest the annual prevalence of SAPHO syndrome varies from 0.00144 in 100,000 in Japanese individuals to fewer than 1 in 10,000 in White individuals. The precise etiopathogenesis of SAPHO remains unclear, but it is generally considered an autoinflammatory syndrome that may be related to various etiologies, such as immune dysfunction, infection and genetic predisposition. Owing to the relapsing–remitting disease course, the goal of management is to improve clinical symptoms and prevent disease progression. Various treatments, including nonsteroidal anti-inflammatory drugs, conventional disease-modifying antirheumatic drugs, bisphosphonates, biologics, and antibiotics, are promising options for alleviating the disease.

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“…The mandible is the most common site for head lesions, which are observed in 10.8% of SAPHO syndrome patients, and skull involvement has been reported in only a few cases [ 4 , 5 ]. However, one study reported osteitis lesions that were identified by bone scintigraphy in 157 patients who met the diagnostic criteria for SAPHO syndrome, and lesions of the neurocranium were observed in 7 (4.5%) patients, in the occipital bone (2.5%), temporal bone (1.3%) and parietal bone (0.6%) [ 6 ]. This report suggested that skull involvement might be overlooked in patients with SAPHO syndrome.…”

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confidence: 99%