以下颌骨骨髓炎为首发症状的SAPHO综合征5例临床分析

doi:10.19438/j.cjoms.2018.06.009

中国口腔颌面外科杂志 ›› 2018, Vol. 16 ›› Issue (6): 528-532.doi: 10.19438/j.cjoms.2018.06.009

以下颌骨骨髓炎为首发症状的SAPHO综合征5例临床分析

高炳菊, 李军, 潘丽珍, 邱宇, 林李嵩

福建医科大学附属第一医院口腔颌面外科,福建医科大学面部整复与重建研究室, 福建省颌面医学中心,福建福州 350005

收稿日期:2018-06-12 出版日期:2018-11-20 发布日期:2019-01-11
通讯作者: 林李嵩,E-mail：dr_lls@163.com
作者简介:高炳菊（1988-）,女,硕士,住院医师,E-mail：244749728@qq.com
基金资助:
福建省自然科学基金（2016J01452）

Clinical analysis of 5 cases of SAPHO syndrome with mandible osteomyelitis as initial presentation

GAO Bing-ju, LI Jun, PAN Li-zhen, QIU Yu, LIN Li-song

Department of Oral and Maxillofacial Surgery, The First Affiliated Hospital of Fujian Medical University, Laboratory of Facial Plastic and Reconstructive Surgery, Fujian Maxillofacial Medical Center. Fuzhou 350005, Fujian Province, China

Received:2018-06-12 Online:2018-11-20 Published:2019-01-11

摘要/Abstract

摘要： 目的: 探讨以下颌骨红肿、疼痛,伴或不伴开口受限为主要临床表现的SAPHO病例的诊断和治疗。方法: 通过回顾我科诊治的5例SAPHO患者的临床资料,结合相关文献报道,分析并讨论SAPHO相关的下颌骨骨髓炎在临床表现、诊断及治疗方面的特点。结果: 以下颌骨红肿、疼痛,开口受限为主要临床表现的SAPHO,与弥漫性硬化性下颌骨骨髓炎类似,极易混淆。治疗上应以保守治疗为主,不提倡手术治疗。结论: SAPHO是一种少见疾病,发生在下颌骨者更是罕见,应加强对此类疾病的认识,掌握其诊断标准,提高早期诊断率,减少不必要的有创操作及手术。

关键词: SAPHO, 下颌骨骨髓炎, 临床表现

Abstract: PURPOSE: To discuss the diagnosis and treatment of SAPHO patients whose main symptoms were painful swelling and inflammation of the mandible, with or without restriction of mouth opening. METHODS: The clinical data of 5 patients with SAPHO in our department were reviewed, clinical manifestations, diagnosis and treatment of SAPHO related osteomyelitis of the mandible were discussed in combination with reported cases. RESULTS: SAPHO patients whose main symptoms were painful swelling and inflammation of the mandible, with or without restriction of mouth opening, were similar to diffuse sclerosing osteomyelitis of the mandible. For such disease, conservative treatment was the main treatment, and surgical treatment was not recommended. CONCLUSIONS: SAPHO is a rare disease, especially on the mandible. We should enhance our understanding of SAPHO and master the diagnostic criteria to improve early diagnosis rate and reduce unnecessary operation.

Key words: SAPHO, Osteomyelitis of mandible, Clinical features

中图分类号:

R782.3

高炳菊, 李军, 潘丽珍, 邱宇, 林李嵩. 以下颌骨骨髓炎为首发症状的SAPHO综合征5例临床分析[J]. 中国口腔颌面外科杂志, 2018, 16(6): 528-532.

GAO Bing-ju, LI Jun, PAN Li-zhen, QIU Yu, LIN Li-song. Clinical analysis of 5 cases of SAPHO syndrome with mandible osteomyelitis as initial presentation[J]. China J Oral Maxillofac Surg, 2018, 16(6): 528-532.

参考文献

[1] Chamot AM, Benhamou CL, Kahn MF, et al.Acne-pustulosis-hyperostosis-osteitis syndrome. Results of a national survey. 85 cases[J]. Rev Rhum Mal Osteoartic, 1987, 54(3): 187-196.
[2] Müller-Richter, UD, Roldán JC, Mörtl M, et al. SAPHO syndrome with ankylosis of the temporomandibular joint[J]. Int J Oral Maxillofac Surg, 2009, 38(12): 1335-1341.
[3] Kahn MF, Hayem F, Fiayem C, et al.Is diffuse sclerosing osteotnyelitis of the mandible part of the synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome?[J]. Oral Surg Oral Med Oral Pathol, 1994, 78(5): 594-598.
[4] Fleuridas G, Teysseres N, Ragot JP, et al.Diffuse sclerosing osteomyelitis of the mandible and SAPHO syndrome[J]. Rev Stomatol Chir Maxillofac, 2002, 103(2): 96-104.
[5] Hayem G, Bouchaud-Chabot A, Benali K, et al.SAPHO syndrome: a long-term follow-up study of 120 cases[J]. Semin Arthritis Rheum, 1999, 29(3): 159-171.
[6] Carneiro S, Sampaio-Barros PD.SAPHO syndrome[J]. Rheum Dis Clin North Am, 2013, 39(2): 401-418.
[7] Kahn MF, Khan MA.The SAPHO syndrome[J]. Baillieres Clin Rheumatol, 1994, 8(2): 333-362.
[8] Kotilainen P, Gullichsen RE, Saario R, et al.Aseptic spondylitis as the initial manifestation of the SAPHO syndrome[J]. Eur Spine J, 1997, 6(5): 327-329.
[9] Hurtado-Nedelec M, Chollet-Martin S, Nicaise-Roland P.Characterization of the immune response in the synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome[J]. Rheumatology(Oxford), 2008, 47(8): 1160-1167.
[10] Assmann G, Simon P.The SAPHO syndrome-are microbes involved?[J]. Best Prac Res Clin Rheumatol, 2011, 25(3): 423-437.
[11] Paparo F, Revelli M, Semprini A, et al.Seronegative spondyloarthropathies: what radiologists should know[J]. Radiol Med, 2014, 119(3): 156-163.
[12] Nguyen MT, Borchers A, Selmi C, et al.The SAPHO syndrome[J]. Semin Arthritis Rheum, 2012, 42(3): 254-265.
[13] Assmann G, Wagner AD, Monika M, et al.Single-nucleotide polymorphisms p53 G72C and Mdm2 T309G in patients with psoriasis, psoriatic arthritis, and SAPHO syndrome[J]. Rheumatol Int, 2010, 30(10): 1273-1276.
[14] Golla A, Jansson A, Ramser J, et al.Chronic recurrent multifocal osteomyelitis (CRMO): evidence for a susceptibility gene located on chromosome 18q21.3-18q22[J]. Eur J Hum Genet, 2002, 10(3): 217-221.
[15] Hurtado-Nedelec M, Chollet-Martin S, Chapeton D, et al.Genetic susceptibility factors in a cohort of 38 patients with SAPHO syndrome: a study of PSTPIP2, NOD2, and LPIN2 genes[J]. J Rheumatol, 2010, 37(2): 401-409.
[16] Queiro R, Moreno P, Sarasqueta C, et al, Synovitis-acne-pustulosis-hyperostosis-osteitis syndrome and psoriatic arthritis exhibit a different immune genetic profile[J]. Clin Exp Rheumatol, 2008, 26(1): 125-128.
[17] Marí A, Morla A, Melero M, et al.Diffuse sclerosing osteomyelitis (DSO) of the mandible in SAPHO syndrome: a novel approach with anti-TNF therapy. Systematic review[J]. J Craniomaxillofac Surg, 2014, 42(8): 1990-1996.
[18] Hayem G.Valuable lessons from SAPHO syndrome[J]. Joint Bone Spine, 2007, 74(2): 123-126.
[19] Suei Y, Taguchi A, Tanimoto K: Diagnosis and classification of mandibular osteomyelitis[J]. Oral Surg Oral Med Oral Pathol Oral Radiol Endod, 2005, 100(2): 207-214.
[20] Olivieri I, Padula A, Palazzi C.Pharmacological management of SAPHO syndrome[J]. Expert Opin Investig Drugs, 2006, 15(10): 1229-1233.
[21] Hayem G, Bouchaud-Chabot A, Benali K, et al.SAPHO syndrome: a long-term follow-up study of 120 cases[J]. Semin Arthritis Rheum, 1999, 29(3): 159-171.
[22] Wagner AD, Andresen J, Jendro MC, et al.Sustained response to tumor necrosis factor alpha-blocking agents in two patients with SAPHO syndrome[J]. Arthritis Rheum, 2002, 46(7): 1965-1968.
[23] Kopterides P, Pikazis D, Koufos C.Successful treatment of SAPHO syndrome with zoledronic acid[J]. Arthritis Rheum, 2004, 50(9): 2970-2973.
[24] Galadari H, Bishop AG, Venna SS, et al.Synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome treated with acombination of isotretinoin and pamidronate[J]. J Am Acad Dermatol, 2009, 6l(1): 123-l25.
[25] Hatano H, Shigeishi H, Higashikawa K, et al.A case of SAPHO syndrome with diffuse sclerosing osteomyelitis of the mandible treated successfully with prednisolone and bisphosphonate[J]. J Oral Maxillofac Surg, 2012, 70(3): 626-631.
[26] Suei Y, Tanimoto K, Miyauchi M, et al.Partial resection of the mandible for the treatment of diffuse sclerosing osteomyelitis: report of four cases[J]. J Oral Maxillofac Surg, 1997, 55(4): 410-415.
[27] Roldán JC, Terheyden H, Dunsche A, et al.Acne with chronic recurrent multifocal osteomyelitis involving the mandible as part of the SAPHO syndrome: case report[J]. Br J Oral Maxillofac Surg, 2001, 39(2): 141-144.
[28] Zemann W, Pau M, Feichtinger M, et al.SAPHO syndrome with affection of the mandible: diagnosis, treatment, and review of literature[J]. Oral Surg Oral Med Oral Pathol Oral Radiol Endod, 2011, 111(2): 190-195.
[29] Moll C, Hernández MV, Cañete JD, et al.Ilium osteitis as the main manifestation of the SAPHO syndrome: response to infliximab therapy and review of the literature[J]. Semin Arthritis Rheum, 2008, 37(5): 299-306.
[30] Reichardt LF, Pitak-Arnnop P, Hemprich A, et al.Chronic refractory osteomyelitis as a part of synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome: a diagnostic challenge[J]. ANZ J Surg, 2011, 81(9): 642-643.

以下颌骨骨髓炎为首发症状的SAPHO综合征5例临床分析

Clinical analysis of 5 cases of SAPHO syndrome with mandible osteomyelitis as initial presentation

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