Background: SAPHO syndrome (synovitis, acne, pustulosis, hyperostosis, osteitis) is both a dermatological and a rheumatological entity, with the common denominator of an aseptic inflammatory process, the bone manifestations of which are the essential element of the diagnosis. Despite its chronic and recurrent nature, SAPHO syndrome is very rarely the cause of major disability, even after several decades of evolution. Objective: We want to illustrate through three clinical cases, the importance of the anamnesis, the clinic, the diagnostic criteria in the diagnosis of SAPHO syndrome. Presentation of the cases: We studied the clinical file of three patients consulted in the Rheumatology Department of the CHU Aristide LeDantec in Dakar, Senegal, between January 2012 and December 2020, suffering from SAPHO syndrome of diagnosis established in accordance with the diagnostic criteria of SAPHO syndrome proposed by Khan et al. of 1994. Conclusion: SAPHO syndrome is a rare pathological entity, over a period of 8 years we report 3 cases.
Freyschmidt, J. and Freyschmidt, G. (1996) Haut-, Schleimhaut- und Skeletterkrankungen SKIBO-Diseases. Springer-Verlag, Berlin, Heidelberg. https://doi.org/10.1007/978-3-642-60991-6
Kahn, M.F. and Khan, M.A. (1994) The SAPHO-Syndrome. Bailliére’s Clinical Rheumatology, 8, 333-362. https://doi.org/10.1016/S0950-3579(94)80022-7
Mari, A., Morla, A., Melero, M., Schiavone, R. and Rodríguez, J. (2014) Diffuse Sclerosing Osteomyelitis (DSO) of the Mandible in SAPHO Syndrome: A Novel Approach with Anti-TNF Therapy. Systematic Review. Journal of Cranio-Maxillo-facial Surgery, 42, 1990-1996. https://doi.org/10.1016/j.jcms.2014.09.004
Golla, A., Jansson, A., Ramser, J., Hellebrand, H., Zahn, R., Meitinger, T., et al. (2002) Chronic Recurrent Multifocal Osteomyelitis (CRMO): Evidence for a Susceptibility Gene Located on Chromosome 18q21.3 18q22. European Journal of Human Genetics, 10, 217-221. https://doi.org/10.1038/sj.ejhg.5200789
Kahn, M.F. (1995) Actualité du syndrome SAPHO. Presse Médicale, 24, 338-340. [In French]
Chamot, A.M. and Kahn, M.F. (1994) Das SAPHO-Syndrom. Zeitschrift für Rheumatologie, 53, 234-242.
Hayem, G., Bouchaud, A., Benali, K., Roux, S., Palazzo, E., Silbermann-Hoffman, O., et al. (1999) SAPHO Syndrome: A Long-Term Follow-up Study of 120 Cases. Seminars in Arthritis and Rheumatism, 29, 159-171. https://doi.org/10.1016/S0049-0172(99)80027-4
Kaiser, H. (1995) Aktuelles über das SAPHO-Syndrom. Zeitschrift für Rheumatologie, 54, 254.
Schilling, F. and Kessler, S. (2000) The SAPHO Syndrome—Clinical and Radiological Differentiation and Classification on the Basis of 86 Cases. Zeitschrift für Rheumatologie, 59, 1-28. https://doi.org/10.1007/s003930050001
Schilling, F. (1998) Das SAPHO-Syndrom—Nosologische Heterogenität und diagnostische Differenzierung—Mit rheumatologischer Osteologie hyperostotischer Spondarthritiden. Aktuelle Rheumatologie, 23, 1-64.
Toussirot, E., Dupond, J.L. and Wendling, D. (1997) Spondylodiscitis in SAPHO Syndrome: A Series of Eight Cases. Annals of the Rheumatic Diseases, 56, 52-58. https://doi.org/10.1136/ard.56.1.52
Van Doornum, S., Barraclough, D., McColl, G. and Wicks, I. (2000) SAPHO: Rare or Just Not Recognized? Seminars in Arthritis and Rheumatism, 30, 70-77. https://doi.org/10.1053/sarh.2000.8371
Bahri, M., Louzir, B. and Orthmani, S. (2013) Le Syndrome SAPHO. http://www.stmi.org.tn/docs/sapho.htm
Schultz, Ch., Holterus, P.M., Seidel, A., Jonas, S., Barthel, M., Kruse, K., et al. (1999) Chronic Recurrent Multifocal Osteomyelitis in Children. The Pediatric Infectious Disease Journal, 18, 1008-1013. https://doi.org/10.1097/00006454-199911000-00015
Hukuda, S., Minami, M., Saito, T., Mitsui, H., Matsui, N., Komatsubara, Y., et al. (2001) Spondyloarthropathies in Japan: Nationwide Questionnaire Survey Performed by the Japan Ankylosing Spondylitis Society. The Journal of Rheumatology, 28, 554-559.
Jurik, A.G., Helmig, O. and Graudal, H. (1988) Skeletal Disease, Arthro-Osteitis, in Adult Patients with Pustulosis Palmoplantaris. Scandinavian Journal of Rheumatology, 70, 3-15. https://doi.org/10.3109/03009748809093406
Sonozaki, H., Kawashima, M., Hongo, O., Yaoita, H., Ikeno, M., Matsuura, M., et al. (1981) Incidence of Arthro-Osteitis in Patients with Pustulosis Palmaris et Plantaris. Annals of the Rheumatic Diseases, 40, 554-557. https://doi.org/10.1136/ard.40.6.554
Szanto, E. and Linse, U. (1991) Arthropathy Associated with Palmoplantar Pustulosis. Clinical Rheumatology, 10, 130-135. https://doi.org/10.1007/BF02207650
Chamot, A.M., Benhamou, C.L., Kahn, M.F., Beraneck, L., Kaplan, G. and Prost, A. (1987) Le syndrome acné-pustulose-hyperostoseostéite (SAPHO). Résultat d’une enquête nationale. Quatre-vingt-cinq observations. Revue du rhumatisme et des maladies osteo-articulaires, 54, 187-196.
Huaux, J.P., Esselinckx, W., Rombouts, J.J., Maldague, B., Malghem, J., Devogelaer, J.P., et al. (1988) Pustulotic Arthro-Osteitis and Chronic Recurrent Multifocal Osteomyelitis in Children. Report of Three Cases. The Journal of Rheumatology, 15, 95-100.
Patterson, A.C. and Bentley-Corbett, K. (1985) Pustulotic Arthroosteitis. The Journal of Rheumatology, 12, 611-614.
Govoni, M., Colina, M., Massara, A. and Trotta, F. (2009) SAPHO Syndrome and Infections. Autoimmunity Reviews, 8, 256-259. https://doi.org/10.1016/j.autrev.2008.07.030
Claude, P., Clerk, D., Cariou, D., Lavabre, C., Veneucie, P.Y. and Bissob, M. (1996) SAPHO Syndromeand Pyoderma Gangrenosum: Is It Fortuitous? The Journal of Rheumatology, 23, 400-402.
Edwards, T.C., Stapleton, B., Bond, M.J. and Barrett, F.F. (1986) Sweet’s Syndrome with Multifocal Sterile Osteomyelitis. American Journal of Diseases of Children, 140, 817-818. https://doi.org/10.1001/archpedi.1986.02140220099042
DiMeco, F., Clatterbuck, R.E., Li, K.W., McCarthy, E.F. and Olivi, A. (2000) Synovitis, Acne, Pus-Tulosis, Hyperostosis, and Osteitis Syndrome Presenting as a Primary Calvariallesion. Case Report and Review of the Literature. Journal of Neurosurgery, 93, 693-697. https://doi.org/10.3171/jns.2000.93.4.0693
Kawai, K., Doita, M., Tateishi, H. and Hirohata, K. (1988) Bone and Joint Lesions Associated with Pustulosis Palmaris and Plantaris. The Journal of Bone and Joint Surgery, 70-B, 117-122. https://doi.org/10.1302/0301-620X.70B1.3276698
Edlund, E., Johnsson, U., Lidgren, L., Pettersson, H., Sturfeld, G., Svensson, B., et al. (1988) Palmoplantar Pustulosis and Sternoclavicular Arthro-Osteitis. Annals of the Rheumatic Diseases, 47, 809-815. https://doi.org/10.1136/ard.47.10.809
Aubry-Rozier, B., Basch, A. and Dudler, J. (2012) Ostéomyélite sclérosante diffuse de la mandibule et SAPHO. Rev Med Interne, 33, e34-e37. https://doi.org/10.1016/j.revmed.2011.07.011
Deltombe, T., Nisolle, J.F., Boutsen, Y., Gustin, T., Gilliard, C. and Hanson, P. (1999) Cervical Spinalcord Injury in SAPHO Syndrome. Spinal Cord, 37, 301-304. https://doi.org/10.1038/sj.sc.3100816
Chamot, A.M., Benhamou, C.L., Kahn, M.F., Beraneck, L., Kaplan, G. and Prost, A. (1987) Acne-Pustulosis Hyperostosis-Osteitis Syndrome. Results of a National Survey. 85 Cases. Revue du rhumatisme et des maladies osteo-articulaires, 3, 187-196.
Collange, C., Brantus, J.F., Sidot, C. and Meunier, P.J. (1996) Sapho Syndrome with Femoral Hyper-Ostosis. Two Case-Reports. Revue du Rhumatisme, 63, 367-370.
Rosner, I.A., Burg, C.G., Wisnieski, J.J., Schacter, B.Z. and Richter, D.E. (1993) The Clinical Spectrum of the Arthropathy Associated with Hidrosadenitis Suppurativa and Acneconglobata. The Journal of Rheumatology, 20, 684-687.
Laasonen, L.S., Karvonen, S.L. and Reunala, T.L. (1994) Bone Disease in Adolescents with Acnefulminans and Severe Cystic Acne: Radiologic and Scintigraphic Findings. American Journal of Roentgenology, 162, 1161-1155. https://doi.org/10.2214/ajr.162.5.8166004
Colina, M., Govoni, M., Orzincolo, C. and Trotta, F. (2009) Clinical and Radiologic Evolution of Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis Syndrome: A Single Centerstudy of a Cohort of 71 Subjects. Arthritis Care & Research, 61, 813-821. https://doi.org/10.1002/art.24540
Fritz, J., Tzaribatchev, N., Claussen, C.D., Carrino, J.A. and Horger, M.S. (2009) Chronic Recurrent Multi-focal Osteomyelitis: Comparison of Whole-Body MR Imaging with Radiography and Correlation with Clinical and Laboratory Data. Radiology, 252, 842-851. https://doi.org/10.1148/radiol.2523081335
Depasquale, R., Kumar, N., Lalam, R.K., Tins, B.J., Tyrell, P.N., Singh, J., et al. (2012) SAPHO: what Radiologists Should Know. Clinical Radiology, 67, 195-206. https://doi.org/10.1016/j.crad.2011.08.014
Parlier-Cuau, C. and Laredo, J.D. (2010) Atteinte vertébrale du SAPHO. Journal de Radiologie, 91, 1068-1078. https://doi.org/10.1016/S0221-0363(10)70151-7
Cabay, J.E., Marcelis, S. and Dondelinger, K.F. (1998) La spondilodiscite inflammatoirecomme manifestation radiologique unique du SAPHO. Journal de Radiologie, 79, 337-340.
Benhamou, C.L., Chamot, A.M. and Kahn, M.F. (1988) Synovitis-Acne-Pustulosis Hyperostosis-Osteomyelitis Syndrome (SAPHO). A New Syndrome among the Spondyloar-Thropathies? Clinical and Experimental Rheumatology, 6, 109-112.
Le Loet, X., Bonnet, B., Thomine, E., et al. (1991) Manifestations ostéo-articulaires de la pustulose palmoplantaire. étude prospective de 15 cas. Presse Médicale, 20, 1307-1311.
Marshall, H., Bromilow, J., Thomas, A.L. and Arden, N.K. (2002) Pamidronate: A Novel Treatment for the SAPHO Syndrome? Rheumatology, 41, 231-233. https://doi.org/10.1093/rheumatology/41.2.231-a
Grassin Delyle, L., Vittecoq, O., Bourdel, A., Duparc, F., Michot, C. and Le Loet, X. (2000) Chronic Destructive Oligoarthritis Associated with Propionibacterium acnes in a Female Patient with Acne Vulgaris: Septic-Reactive Arthritis? Arthritis & Rheumatism, 43, 2843-2847. https://doi.org/10.1002/1529-0131(200012)43:12%3C2843::AID-ANR26%3E3.0.CO;2-2
Pillon, P., Pajon, A., Juvin, R., Gaudin, P., Tourne, Y., Pasquier, B., et al. (1992) Hyperostose tibiale et Propionibacterium acnes. Revue du rhumatisme et des maladies osteo-articulaires, 59, 349-351.
Schaeverbeke, T., Lequen, L., de Barbeyrac, B., Labbé, L., Bébéar, C.M., Morrier, Y., et al. (1998) Propionibacterium Acnes Isolated from Synovial Tissue and Fluid in a Patient with Oligoarthritis Associated with Acne and Pustulosis. Arthritis & Rheumatism, 41, 1889-1893. https://doi.org/10.1002/1529-0131(199810)41:10%3C1889::AID-ART23%3E3.0.CO;2-F
Wagner, A.D., Mai, U., Hammer, M. and Zeidler, H. (1997) Longterm Antibiotic Therapy in Patients with SAPHO. Arthritis & Rheumatology, 40, S62 (résumé).
Ballara, S.C., Siraj, Q.H., Maini, R.N. and Venables, P.J. (1999) Sustained Response to Doxycycline Therapy in Two Patients with SAPHO Syndrom. Arthritis & Rheumatism, 42, 818-823. https://doi.org/10.1002/1529-0131(199904)42:4%3C818::AID-ANR29%3E3.0.CO;2-G
Yamamoto, T., Kimura, K., Katayama, I. and Nishioka, K. (1995) Successful Treatment of Severe Arthralgia Associated with Palmoplantar Pustulosis with Low-Dose oral Cyclosporine A. The Journal of Dermatology, 22, 512-514. https://doi.org/10.1111/j.1346-8138.1995.tb03435.x
Rothschild, B., Schils, J. and Lavelle, H. (2000) Potential Therapeutic Approach to SAPHO. Seminars in Arthritis and Rheumatism, 29, 332-334. https://doi.org/10.1016/S0049-0172(00)80020-7
Job-Deslandre, C., Krebs, S. and Kahan, A. (2001) Chronic Recurrent Multifocal Osteomyelitis: Five-Year Outcomes in 14 Pediatric Cases. Joint Bone Spine, 68, 245-251. https://doi.org/10.1016/S1297-319X(01)00276-7
Olivieri, I., Padula, A., Ciancio, G., Salvarani, C., Niccoli, L. and Cantini, F. (2002) Successful Treatment of SAPHO Syndrome with Infliximab: Report of Two Cases. Annals of the Rheumatic Diseases, 61, 375-376. https://doi.org/10.1136/ard.61.4.375
Wagner, A.D., Andresen, J., Jendro, M.C., Hulsemann, J.L. and Zeidler, H. (2002) Sustained Response to Tumor Necrosis Factor Alpha-Blocking Agents in Two Patients with SAPHO Syndrome. Arthritis & Rheumatism, 46, 1965-1968. https://doi.org/10.1002/art.10539
Schilling, F., Palme, E., Stollenwert, R. and Kreitner, K.F. (1997) Chronic Recurrent Multifocal Osteomyelitis (CRMO). Case Report on Adult CRMO with Palmoplantar Pustulosis (Ppp). Aktuelle Rheumatologie, 22, 55-65. https://doi.org/10.1055/s-2008-1043630
van Holsbeek, M., Martel, W., Dequeker, J., Favril, A., Gielen, J., Verschakelen, J., et al. (1989) Soft Tissue Involvment, Mediastinal Pseudotumor, and Venous Thrombosis in Pustulotic Arthro-Osteitis. Skeletal Radiology, 18, 1-8. https://doi.org/10.1007/BF00366761
Schilling, F., Fedlmeier, M., Eckardt, A. and Kessler, S. (2002) Vertebral Manifestation of Chronic Recurrent Multifocal Osteomyelitis (CRMO). Fortschr Röntgenstr, 174, 1236-1242. https://doi.org/10.1055/s-2002-34565
Uhl, M., Leichsenring, M. and Krempien, B. (1995) Chronisch Rezidivierende Multifokale Osteomyelitis. Fortschr Röntgenstr, 162, 527-530.
Campillo, J.F. and Garcia-Pachon, E. (2001) Pleural Effusion Associated with the SAPHO Syndrome. Chest, 120, 1752 (letter). https://doi.org/10.1378/chest.120.5.1752
Legoupil, N., Révelon, G., Allain, J., Voisin, M.C., Rahmouni, A., Chevalier, X., et al. (2001) Iliac Vein Thrombosis Complicating SAPHO Syndrome: MRI and Histological Features of Soft Tissue Lesions. Joint Bone Spine, 68, 79-83. https://doi.org/10.1016/S1297-319X(00)00229-3
Schilling, F. and Märker-Hermann, E. (2003) Die Assoziation der CRMO mit chronisch entzündlichen Darmleiden beim SAPHO-Syndrom—5 eigene Fälle.