Synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome with purely osteolytic, not osteosclerotic, lesions mimicking a malignant tumor.
Synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome with purely osteolytic, not osteosclerotic, lesions mimicking a malignant tumor.
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滑膜炎、痤疮、脓疱病、骨质增生和骨炎综合征，伴有纯粹的溶骨性而非骨硬化性病变，类似于恶性肿瘤。
DOI：
10.1155/2020/6316921
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发表时间：
2020
期刊：
影响因子：
--
通讯作者：
Yonemoto T.
中科院分区：
文献类型：
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作者：
Kinoshita H;Ishii T;Kamoda H;Hagiwara Y;Tsukanishi T;Orita S;Inage K;Hirosawa N;Ohtori S;Yonemoto T.
滑膜炎、痤疮、脓疱病、骨质增生和骨炎（SAPHO）综合征是一种罕见的具有多种表型的炎性疾病。该综合征具有可识别的放射学特征，这在诊断时是最重要的。诊断为SAPHO综合征的病例的X射线显示硬化病变或混合溶解性和硬化病变。SAPHO综合征中的纯溶骨性病变很少见，据我们所知，没有研究报告纯溶骨性病变随着时间的推移会发生骨硬化病变的放射学变化。在此，我们报告的情况下，妇女经历严重的左大腿急性疼痛和病史的掌跖脓疱病。虽然SAPHO综合征被怀疑是因为掌跖脓疱病，根据放射学结果，骨转移的恶性肿瘤或慢性细菌性骨髓炎，由于纯粹的溶骨性病变被怀疑。然而，穿刺活检显示没有恶性肿瘤和细菌培养阴性，因此建议SAPHO综合征。给予非甾体抗炎药、双膦酸盐和皮质类固醇，改善了左大腿疼痛。此外，随着时间的推移，溶骨性病变到骨质疏松病变的放射学变化得到证实，导致SAPHO综合征的诊断。我们的病例表明，不典型的影像学表现的知识是必要的诊断初始SAPHO综合征。
Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare inflammatory disorder with multiple phenotypes. The syndrome has identifiable radiologic characteristics that are the most important when making a diagnosis. X‐rays of cases diagnosed with SAPHO syndrome reveal sclerotic lesions or mixed lytic and sclerotic lesions. Pure osteolytic lesions in SAPHO syndrome are rare, and to the best of our knowledge, no study has reported the radiologic change of purely osteolytic lesions to osteosclerotic lesions over time. Herein, we report on the case of a woman experiencing severe left thigh acute pain and having a medical history of palmoplantar pustulosis. Although SAPHO syndrome was suspected because of palmoplantar pustulosis, based on radiologic findings, bone metastasis of a malignant tumor or chronic bacterial osteomyelitis owing to a purely osteolytic lesion was suspected. However, needle biopsy revealed no malignancy and bacterial culture was negative, thus suggesting SAPHO syndrome. Nonsteroidal anti‐inflammatory drugs, bisphosphonates, and corticosteroids were administered, which improved the left thigh pain. Furthermore, the radiologic change of osteolytic lesions to osteosclerotic lesions over time was confirmed, leading to the diagnosis of SAPHO syndrome. Our case demonstrates that knowledge of atypical radiologic findings is necessary to diagnose initial SAPHO syndrome.