Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis Syndrome with Purely Osteolytic, Not Osteosclerotic, Lesions Mimicking a Malignant Tumor
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 sclero...
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| Format: | Article |
| Language: | English |
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Wiley
2020-01-01
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| Series: | Case Reports in Rheumatology |
| Online Access: | http://dx.doi.org/10.1155/2020/6316921 |
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| author | Hideyuki Kinoshita Takeshi Ishii Hiroto Kamoda Yoko Hagiwara Toshinori Tsukanishi Sumihisa Orita Kazuhide Inage Naoya Hirosawa Seiji Ohtori Tsukasa Yonemoto |
| author_facet | Hideyuki Kinoshita Takeshi Ishii Hiroto Kamoda Yoko Hagiwara Toshinori Tsukanishi Sumihisa Orita Kazuhide Inage Naoya Hirosawa Seiji Ohtori Tsukasa Yonemoto |
| author_sort | Hideyuki Kinoshita |
| collection | DOAJ |
| description | 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. |
| format | Article |
| id | doaj-art-d599a7a4ae914a64804a9a64f6a96e6c |
| institution | Kabale University |
| issn | 2090-6889 2090-6897 |
| language | English |
| publishDate | 2020-01-01 |
| publisher | Wiley |
| record_format | Article |
| series | Case Reports in Rheumatology |
| spelling | doaj-art-d599a7a4ae914a64804a9a64f6a96e6c2025-02-03T01:27:04ZengWileyCase Reports in Rheumatology2090-68892090-68972020-01-01202010.1155/2020/63169216316921Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis Syndrome with Purely Osteolytic, Not Osteosclerotic, Lesions Mimicking a Malignant TumorHideyuki Kinoshita0Takeshi Ishii1Hiroto Kamoda2Yoko Hagiwara3Toshinori Tsukanishi4Sumihisa Orita5Kazuhide Inage6Naoya Hirosawa7Seiji Ohtori8Tsukasa Yonemoto9Department of Orthopedic Surgery, Chiba Cancer Center, 666-2 Nitonacho, Chuo-ku, Chiba 260-8717, JapanDepartment of Orthopedic Surgery, Chiba Cancer Center, 666-2 Nitonacho, Chuo-ku, Chiba 260-8717, JapanDepartment of Orthopedic Surgery, Chiba Cancer Center, 666-2 Nitonacho, Chuo-ku, Chiba 260-8717, JapanDepartment of Orthopedic Surgery, Chiba Cancer Center, 666-2 Nitonacho, Chuo-ku, Chiba 260-8717, JapanDepartment of Orthopedic Surgery, Chiba Cancer Center, 666-2 Nitonacho, Chuo-ku, Chiba 260-8717, JapanDepartment of Orthopaedic Surgery, Graduate School of Medicine, Chiba University, 1-8-1 Inohana, Chuo-ku, Chiba 260-8670, JapanDepartment of Orthopaedic Surgery, Graduate School of Medicine, Chiba University, 1-8-1 Inohana, Chuo-ku, Chiba 260-8670, JapanDepartment of Orthopaedic Surgery, Graduate School of Medicine, Chiba University, 1-8-1 Inohana, Chuo-ku, Chiba 260-8670, JapanDepartment of Orthopaedic Surgery, Graduate School of Medicine, Chiba University, 1-8-1 Inohana, Chuo-ku, Chiba 260-8670, JapanDepartment of Orthopedic Surgery, Chiba Cancer Center, 666-2 Nitonacho, Chuo-ku, Chiba 260-8717, JapanSynovitis, 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.http://dx.doi.org/10.1155/2020/6316921 |
| spellingShingle | Hideyuki Kinoshita Takeshi Ishii Hiroto Kamoda Yoko Hagiwara Toshinori Tsukanishi Sumihisa Orita Kazuhide Inage Naoya Hirosawa Seiji Ohtori Tsukasa Yonemoto Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis Syndrome with Purely Osteolytic, Not Osteosclerotic, Lesions Mimicking a Malignant Tumor Case Reports in Rheumatology |
| title | Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis Syndrome with Purely Osteolytic, Not Osteosclerotic, Lesions Mimicking a Malignant Tumor |
| title_full | Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis Syndrome with Purely Osteolytic, Not Osteosclerotic, Lesions Mimicking a Malignant Tumor |
| title_fullStr | Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis Syndrome with Purely Osteolytic, Not Osteosclerotic, Lesions Mimicking a Malignant Tumor |
| title_full_unstemmed | Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis Syndrome with Purely Osteolytic, Not Osteosclerotic, Lesions Mimicking a Malignant Tumor |
| title_short | Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis Syndrome with Purely Osteolytic, Not Osteosclerotic, Lesions Mimicking a Malignant Tumor |
| title_sort | synovitis acne pustulosis hyperostosis and osteitis syndrome with purely osteolytic not osteosclerotic lesions mimicking a malignant tumor |
| url | http://dx.doi.org/10.1155/2020/6316921 |
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