于梅艳;张亚男;任蒙蒙;洪月颖;李俊秋;李忱;卢新宇;张文;霍健伟;

• 1:首都医科大学附属北京中医医院放射科
• 2:北京协和医院中医科
• 3:中国医学科学院北京协和医院临床医学部
• 4:北京协和医院风湿免疫科

摘要(Abstract)：

目的分析SAPHO综合征颌骨受累的磁共振表现,提高各科医师对此疾病的认识。材料与方法回顾性分析9例SAPHO综合征颌骨MRI图像,由两名放射科医师共同阅片,评估颌骨骨髓水肿、骨质肥厚、脂质沉积的范围及程度;下颌关节、周围组织的受累范围;病变的扩散受限情况;有争议处通过讨论达成一致。结果本组9例SAPHO综合征,8例为儿童/青少年。磁共振表现为不同程度的下颌骨受累,主要累及单侧下颌支及下颌角,并邻近体部,上颌骨未见受累。9例均出现骨髓水肿,其中8例不对称。7例合并骨质肥厚,均为髓质部受累。5例合并周围软组织水肿,其中1例累及邻近肌群,9例均出现周围多发淋巴结。1例行扩散加权成像检查,未见明确扩散受限。结论磁共振可明确显示SAPHO综合征颌骨受累病变范围,主要表现为下颌骨不对称骨质肥厚伴水肿,可累及邻近软组织,伴多发淋巴结,为临床治疗、随访提供帮助。

关键词(KeyWords)： SAPHO综合征;磁共振成像;颌骨;病例报道

Abstract:

Keywords:

基金项目(Foundation): 中国医学科学院医学与健康科技创新工程项目(编号：2017-I2M-3-001)~~

作者(Author): 于梅艳;张亚男;任蒙蒙;洪月颖;李俊秋;李忱;卢新宇;张文;霍健伟;

Email:

DOI:

参考文献(References)：

• [1]Chamot AM,Benhamou CL,Kahn MF,et al.Acne-pustulosishyperostosisosteitis syndrome.Results of a national survey.85 cases.Rev Rhum Mal Osteoartic,1987,54(3):187-196.
• [2]Jurik AG,Klicman RF,Simoni P,et al.SAPHO and CRMO:The value of imaging.Semin Musculoskelet Radiol,2018,22(2):207-224.DOI:10.1055/s-0038-1639469.
• [3]Cao Y,Li C,Yang Q,et al.Three patterns of osteoarticular involvement in SAPHO syndrome:a cluster analysis based on whole body bone scintigraphy of 157 patients.Rheumatology (Oxford),2019,58(6):1047-1055.DOI:10.1093/rheumatology/key415.
• [4]Wang Y,Yang C,Zhang W,et al.Monofocal chronic nonbacterial osteomyelitis in the mandible accompanied with mucocutaneous disease.J Craniofac Surg,2017,28(6):547-551.DOI:10.1097/SCS.0000000000003840.
• [5]Andre CV,Khonsari RH,Ernenwein D,et al.Osteomyelitis of the jaws:A retrospective series of 40 patients.J Stomatol Oral Maxillofac Surg,2017,118(5):261-264.DOI:10.1016/j.jormas.2017.04.007.
• [6]Gao BJ,Li J,Pan LZ,et al.Clinical analysis of 5 cases of SAPHOsyndrome with mandible osteomyelitis as initial presentation.Chin J Oral&Maxillofac Surg,2018,16(6):528-532.DOI:10.19438/j.cjoms.2018.06.009.高炳菊,李军,潘丽珍,等.以下颌骨骨髓炎为首发症状的SAPHO综合征5例临床分析.中国口腔颌面外科杂志,2018,16(6):528-532.DOI:10.19438/j.cjoms.2018.06.009.
• [7]Nguyen MT,Borchers A,Selmi C,et al.The SAPHO syndrome.Semin Arthritis Rheum,2012,42(3):254-265.DOI:10.1016/j.semarthrit.2012.05.006.
• [8]Wang M,Li Y,Cao Y,Lu X,et al.Mandibular involvement in SAPHOsyndrome:a retrospective study.Orphanet J Rare Dis,2020,151(1):312-321.DOI:10.1186/s13023-020-01589-0.
• [9]Baltensperger M,Gratz K,Bruder E,et al.Is primary chronic osteomyelitis a uniform disease?Proposal of a classification based on a retrospective analysis of patients treated in the past 30 years.JCranio Maxillofac Facial Surg,2004,32(1):43-50.DOI:10.1016/j.jcms.2003.07.008.
• [10]Obel G,Krogdahl A,Thygesen T,et al.Juvenile mandibular chronic osteomyelitis:3 cases and a literature review.J Oral Maxillofac Surg,2013,71(2):305-309.DOI:10.1016/j.joms.2012.02.013.
• [11]Suei Y,Taguchi A,Tanimoto K.Diffuse sclerosing osteomyelitis of the mandible:its characteristics and possible relationship to synovitis,acne,pustulosis,hyperostosis,osteitis (SAPHO) syndrome.J Oral Maxillofac Surg,1996,54(10):1194-1199.DOI:10.1016/s0278-2391(96)90349-3.
• [12]Bhat CS,Anderson C,Harbinson A,et al.Chronic non bacterial osteitis-a multicentre study.Pediatr Rheumatol Online J,2018,16(1):74.DOI:10.1186/s12969-018-0290-5.
• [13]Hofmann SR,Kapplusch F,Girschick HJ,et al.Chronic recurrent multifocal osteomyelitis (CRMO):Presentation,pathogenesis,and treatment.Curr Osteoporos Rep,2017,15(6):542-554.DOI:10.1007/s11914-017-0405-9.
• [14]Fleuridas G,Teysseres N,Ragot JP,et al.Diffuse sclerosing osteomyelitis of the mandible and SAPHO syndrome.Rev Stomatol Chir Maxillofac,2002,103(2):96-104.DOI:10.1016/j.revmed.2011.07.011.
• [15]Khanna G,Sato TS,Ferguson P.Imaging of chronic recurrent multifocal osteomyelitis.Radiographics,2009,29(4):1159-1177.DOI:10.1148/rg.294085244.
• [16]Kotaki S,Gamoh S,Yoshida H,et al.SAPHO syndrome of the temporomandibular joint associated with trismus:a case report and review of the literature.Oral radiology,2020,36(2):197-202.DOI:10.1007/s11282-019-00405-1.
• [17]Long L,He HY,Zhang P.Osteomyelitis of jaw as the main manifestation of SAPHO syndrome:Report of one case and review of the literature.Chin J Oral&Maxillofac Surg,2010,8(6):570-573.DOI:1672-3244(2010)06-0570-04.龙丽,何慧英,张萍.SAPHO综合征累及颌骨:1例报告及文献复习.中国口腔颌面外科杂志,2010,8(6):570-573.DOI:1672-3244(2010)06-0570-04.
• [18]Kikuchi T,Fujii H,Fujita A,et al.Mandibular osteitis leading to the diagnosis of SAPHO syndrome.Case Reports in Radiol,2018,2018:9142362.DOI:10.1155/2018/9142362.
• [19]Zhang YN,Li JQ,Wen QX,et al.Magnetic resonance imaging evaluation of axial bone in four cases with SAPHO syndrome.Chin JMagn Reson Imaging,2018,9(11):836-841.DOI:10.12015/issn.1674-8034.2018.11.007.张亚男,李俊秋,温庆祥,等.四例SAPHO综合征患者中轴骨的磁共振评估.磁共振成像,2018,9(11):836-841.DOI:10.12015/issn.1674-8034.2018.11.007
• [20]El Mortaji H,Elghazi M,Belhadj Z,et al.Aneurysmal bone cyst of the ethmoid on fibrous dysplasia:A usual association within a rare location.Radiol Case Rep,2019,1411(11):1356-1359.DOI:10.1016/j.radcr.2019.07.015.
• [21]Ta NH,Addison A,Beigi B,et al.Unilateral visual loss resulting from orbital encroachment of an ethmoidal juvenile trabecular ossifying fibroma.Ann R Coll Surg Engl,2019,1014(4):e111-e114.DOI:10.1308/rcsann.2019.0030.
• [22]Vo Quang S,Sicard L,Samama M,et al.Mandibular lymphoma.JStomatol Oral Maxillofac Surg,2018,1191(1):49-51.DOI:10.1016/j.jormas.2017.10.004.
• [23]Ogura I,Sasaki Y,Kameta A,et al.A rare case of malignant melanoma of the mandible:CT and MRI findings.Chin J Dent Res,2017,20(2):111-114.DOI:10.3290/j.cjdr.a38276.
• [24]Kim JE,Yi WJ,Heo MS,et al.Langerhans cell histiocytosis of the jaw,a mimicker of osteomyelitis on CT and MR images:A retrospective analysis.Medicine (Baltimore),2019,98(27):e16331.DOI:10.1097/MD.0000000000016331.