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Dramatic response of synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome to tofacitinib monotherapy: a case report
The synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare condition. Its treatment remains a challenge for clinicians, and often yields mixed results. We report the case of a 51-year-old Caucasian woman who presented with SAPHO syndrome with mainly axial involvement. She...
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Published in: | Journal of medical case reports 2024-02, Vol.18 (1), p.57-57, Article 57 |
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description | The synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare condition. Its treatment remains a challenge for clinicians, and often yields mixed results.
We report the case of a 51-year-old Caucasian woman who presented with SAPHO syndrome with mainly axial involvement. She had been treated with sulfasalazine and anti-inflammatory drugs for many years without any success. A few weeks after starting treatment with tofacitinib, both clinical and biological parameters dramatically improved. Imaging also showed considerable regression of the vertebral and pelvic lesions. However, tofacitinib had to be discontinued due to the occurrence of pulmonary embolism. Consequently, recurrence of bone pain and biologic inflammation was rapidly observed.
Anti-JAKs are an interesting treatment option in the management of SAPHO syndrome that need further clinical trials and assessment for validating response. |
doi_str_mv | 10.1186/s13256-024-04366-w |
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We report the case of a 51-year-old Caucasian woman who presented with SAPHO syndrome with mainly axial involvement. She had been treated with sulfasalazine and anti-inflammatory drugs for many years without any success. A few weeks after starting treatment with tofacitinib, both clinical and biological parameters dramatically improved. Imaging also showed considerable regression of the vertebral and pelvic lesions. However, tofacitinib had to be discontinued due to the occurrence of pulmonary embolism. Consequently, recurrence of bone pain and biologic inflammation was rapidly observed.
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We report the case of a 51-year-old Caucasian woman who presented with SAPHO syndrome with mainly axial involvement. She had been treated with sulfasalazine and anti-inflammatory drugs for many years without any success. A few weeks after starting treatment with tofacitinib, both clinical and biological parameters dramatically improved. Imaging also showed considerable regression of the vertebral and pelvic lesions. However, tofacitinib had to be discontinued due to the occurrence of pulmonary embolism. Consequently, recurrence of bone pain and biologic inflammation was rapidly observed.
Anti-JAKs are an interesting treatment option in the management of SAPHO syndrome that need further clinical trials and assessment for validating response.</description><subject>Acne</subject><subject>Anti-inflammatory agents</subject><subject>Arthritis</subject><subject>Bisphosphonates</subject><subject>Blood tests</subject><subject>Case report</subject><subject>Case reports</subject><subject>Disodium pamidronate</subject><subject>Exostosis</subject><subject>JAK-inhibitors</subject><subject>Kinases</subject><subject>Magnetic resonance imaging</subject><subject>Nonsteroidal anti-inflammatory drugs</subject><subject>Patients</subject><subject>Pelvis</subject><subject>Pulmonary embolism</subject><subject>Reimbursement</subject><subject>Rheumatic diseases</subject><subject>Sacroiliitis</subject><subject>SAPHO syndrome</subject><subject>Skin</subject><subject>Sulfasalazine</subject><subject>Synovitis</subject><subject>TNF inhibitors</subject><subject>Tofacitinib</subject><subject>Tomography</subject><subject>Tumor necrosis factor-TNF</subject><subject>Vertebrae</subject><issn>1752-1947</issn><issn>1752-1947</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2024</creationdate><recordtype>article</recordtype><sourceid>PIMPY</sourceid><sourceid>DOA</sourceid><recordid>eNptUstu1TAQjRCIlsIPsECW2LBoil9xHHZVoVCpEhtYWxNn3PoqiYOdtLorfh2nactDyJbsGZ1zZsY-RfGa0RPGtHqfmOCVKimXJZVCqfL2SXHI6oqXrJH10z_uB8WLlHaUVko34nlxILRQVDN1WPz8GGGA2VsSMU1hTEiCI2k_hhs_-3RMwI54TKYlzUsf0pq53k8YQ5q3CMaO5ABX9MrrYhiQzCFvBzZnR9-SIYxhvsYI0_4DAWIhl4k4hTi_LJ456BO-uj-Piu_nn76dfSkvv36-ODu9LK2smrnUFUhLrWAKamZBURCKcWprbFVbo624Eo4L5qBTjjpZsxqoRgZWUykpiqPiYtPtAuzMFP0AcW8CeHOXCPHKQMzP0KNRrstartZcVBKhAwHUMWgtMtlU3ar1btOaYvixYJrN4JPFvocRw5IMb7jmkilJM_TtP9BdWOKYJ82o_Bt1o6T-jbqCXN-PLswR7CpqTnMbNadCVRl18h9UXh0O3oYRnc_5vwh8I9j8XSmie5ybUbM6yGwOMtlB5s5B5jaT3tx3vLQDdo-UB8uIX6q_wg8</recordid><startdate>20240216</startdate><enddate>20240216</enddate><creator>Dierckx, Stéphanie</creator><creator>Nisolle, Jean-François</creator><creator>Boutsen, Yves</creator><general>BioMed Central Ltd</general><general>BioMed Central</general><general>BMC</general><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7RV</scope><scope>7X7</scope><scope>7XB</scope><scope>88C</scope><scope>88E</scope><scope>8C1</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>KB0</scope><scope>M0S</scope><scope>M0T</scope><scope>M1P</scope><scope>NAPCQ</scope><scope>PIMPY</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>7X8</scope><scope>DOA</scope><orcidid>https://orcid.org/0000-0002-9062-679X</orcidid></search><sort><creationdate>20240216</creationdate><title>Dramatic response of synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome to tofacitinib monotherapy: a case report</title><author>Dierckx, Stéphanie ; 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Its treatment remains a challenge for clinicians, and often yields mixed results.
We report the case of a 51-year-old Caucasian woman who presented with SAPHO syndrome with mainly axial involvement. She had been treated with sulfasalazine and anti-inflammatory drugs for many years without any success. A few weeks after starting treatment with tofacitinib, both clinical and biological parameters dramatically improved. Imaging also showed considerable regression of the vertebral and pelvic lesions. However, tofacitinib had to be discontinued due to the occurrence of pulmonary embolism. Consequently, recurrence of bone pain and biologic inflammation was rapidly observed.
Anti-JAKs are an interesting treatment option in the management of SAPHO syndrome that need further clinical trials and assessment for validating response.</abstract><cop>England</cop><pub>BioMed Central Ltd</pub><pmid>38360816</pmid><doi>10.1186/s13256-024-04366-w</doi><tpages>1</tpages><orcidid>https://orcid.org/0000-0002-9062-679X</orcidid><oa>free_for_read</oa></addata></record> |
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subjects | Acne Anti-inflammatory agents Arthritis Bisphosphonates Blood tests Case report Case reports Disodium pamidronate Exostosis JAK-inhibitors Kinases Magnetic resonance imaging Nonsteroidal anti-inflammatory drugs Patients Pelvis Pulmonary embolism Reimbursement Rheumatic diseases Sacroiliitis SAPHO syndrome Skin Sulfasalazine Synovitis TNF inhibitors Tofacitinib Tomography Tumor necrosis factor-TNF Vertebrae |
title | Dramatic response of synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome to tofacitinib monotherapy: a case report |
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