Advertisement

SAPHO syndrome of the temporomandibular joint associated with trismus: a case report and review of the literature

  • Shinya KotakiEmail author
  • Shoko Gamoh
  • Hiroaki Yoshida
  • Takaaki Noma
  • Kaname Tsuji
  • Hironori Akiyama
  • Yoritaka Yotsui
  • Tomio Iseki
  • Kimishige Shimizutani
Case Report
  • 16 Downloads

Abstract

Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a disorder characterized by pustular skin lesions and osteoarticular lesions. Mandibular involvement occurs in approximately 10% of the cases, and is often seen as recurrent mandibular osteitis with bone sclerosis, mainly involving the body of the mandible in the head and neck region. Middle cranial base with temporomandibular joint (TMJ) involvement in SAPHO syndrome can be diagnostically challenging because of its rarity. Herein, we present a case of a 37-year-old man who suffered from trismus and dull pain in the left TMJ region. The initial panoramic image revealed spotty osteolysis around the left condylar head. Computed tomography (CT) images showed an osteosclerotic change in the middle cranial base including the TMJ. Magnetic resonance images showed a cortical bone change in the left TMJ without anterior disk displacement, with spotty low signal intensity in the left condyle bone marrow on T2- weighted images. Our initial diagnosis was osteomyelitis of the middle cranial base including the TMJ region. However, antimicrobial therapy, training for TMJ opening, and a surgical procedure were not effective. A detailed medical interview, careful check for skin lesions, and further imaging examinations including bone scintigraphy and chest CT led to the diagnosis of SAPHO syndrome. The possibility of SAPHO syndrome should be considered in patients suspected of osteomyelitis of the middle cranial base including the TMJ with unknown etiology.

Keywords

SAPHO syndrome TMJ TMD Trismus Bisphosphonate 

Notes

Acknowledgements

This study received funding from the Japan Society for the Promotion of Science (JSPS KAKENHI Grant Number 18K17207). The authors thank Alison Sherwin, PhD, from Edanz Group (www.edanzediting.com/ac) for editing a draft of this manuscript and Shogo Takahashi for creating a figure of this manuscript.

Compliance with ethical standards

Conflict of interest

Each author certifies that he or she has no commercial associations that might pose a conflict of interest in connection with this study.

Human rights statements

All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation (institutional and national) and with the Helsinki Declaration of 1964 and later versions.

Informed consent

Written informed consent for the case to be published (including images, case history and data) was obtained from the patient for publication of this case report, including accompanying images.

References

  1. 1.
    Chamot AM, Benhamou CL, Kahn MF, Beraneck L, Kaplan G, Prost A. Acne-pustulosis-hyperostosis-osteitis syndrome results of a national survey. 85 cases. Rev Rhum Mal Osteoartic. 1987;54(3):187–96 [article in French].Google Scholar
  2. 2.
    Aljuhani F, Tournadre A, Tatar Z, Couderc M, Mathieu S, Malochet-Guinamand S, Soubrier M, Dubost JJ. The SAPHO syndrome: a single-center study of 41 adult patients. J Rheumatol. 2015;42(2):329–34.CrossRefGoogle Scholar
  3. 3.
    Kikuchi T, Fujii H, Fujita A, Sugiyama T, Sugimoto H. Mandibular osteitis leading to the diagnosis of SAPHO syndrome. Case Rep Radiol. 2018;2018:9142362.Google Scholar
  4. 4.
    Marsot-Dupuch K, Doyen JE, Grauer WO, de Givry SC. SAPHO syndrome of the temporomandibular joint associated with sudden deafness. AJNR Am J Neuroradiol. 1999;20(5):902–5.Google Scholar
  5. 5.
    McPhillips A, Wolford LM, Rodrigues DB. SAPHO syndrome with TMJ involvement: review of the literature and case presentation. Int J Oral Maxillofac Surg. 2010;39(12):1160–7.CrossRefGoogle Scholar
  6. 6.
    Kodama Y, Tanaka R, Kurokawa A, Ohnuki H, Sultana S, Hayashi T, Iizuka T, Takagi R. Severe destruction of the temporomandibular joint with complete resorption of the condyle associated with synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome. Oral Surg Oral Med Oral Pathol Oral Radiol. 2013;116(2):e128–e133133.CrossRefGoogle Scholar
  7. 7.
    Benhamou CL, Chamot AM, Kahn MF. Synovitis-acne-pustulosis hyperostosis-osteomyelitis syndrome (SAPHO). A new syndrome among the spondyloarthropathies? Clin Exp Rheumatol. 1988;6(2):109–12.Google Scholar
  8. 8.
    Kahn MF, Hayem F, Hayem G, Grossin M. Is diffuse sclerosing osteomyelitis of the mandible part of the synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome? Analysis of seven cases. Oral Surg Oral Med Oral Pathol. 1994;78(5):594–8.CrossRefGoogle Scholar
  9. 9.
    Eyrich GK, Langenegger T, Bruder E, Sailer HF, Michel BA. Diffuse chronic sclerosing osteomyelitis and the synovitis, acne, pustolosis, hyperostosis, osteitis (SAPHO) syndrome in two sisters. Int J Oral Maxillofac Surg. 2000;29(1):49–53.CrossRefGoogle Scholar
  10. 10.
    Utumi ER, Oliveira Sales MA, Shinohara EH, Takahashi A, Coracin FL, Rocha RG, Paraíso Cavalcanti MG. SAPHO syndrome with temporomandibular joint ankylosis: clinical, radiological, histopathological, and therapeutical correlations. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008;105(3):e67–72.CrossRefGoogle Scholar
  11. 11.
    Müller-Richter UD, Roldán JC, Mörtl M, Behr M, Reichert TE, Driemel O. SAPHO syndrome with ankylosis of the temporomandibular joint. Int J Oral Maxillofac Surg. 2009;38(12):1335–411.CrossRefGoogle Scholar
  12. 12.
    Yanamoto S, Kawasaki G, Yoshitomi I, Mizuno A. Diffuse chronic sclerosing osteomyelitis of the mandible with synovitis, acne, pustulosis, hyperostosis, and osteitis: report of a long-term follow-up case. J Oral Maxillofac Surg. 2010;68(1):212–27.CrossRefGoogle Scholar
  13. 13.
    Okochi K, Kretapirom K, Sumi Y, Kurabayashi T. Longitudinal MRI follow-up of rheumatoid arthritis in the temporomandibular joint: importance of synovial proliferation as an early-stage sign. Oral Radiol. 2011;27(1):83–6.CrossRefGoogle Scholar
  14. 14.
    Kretapirom K, Okochi K, Nakamura S, Tetsumura A, Ohbayashi N, Yoshino N, Kurabayashi T. MRI characteristics of rheumatoid arthritis in the temporomandibular joint. Dentomaxillofac Radiol. 2013;42(4):31627230.CrossRefGoogle Scholar
  15. 15.
    Amital H, Applbaum YH, Aamar S, Daniel N, Rubinow A. SAPHO syndrome treated with pamidronate: an open-label study of 10 patients. Rheumatol. 2004;43(5):658–61.CrossRefGoogle Scholar
  16. 16.
    Delattre E, Guillot X, Godfrin-Valnet M, Prati C, Wendling D. SAPHO syndrome treatment with intravenous pamidronate. Retrospective study of 22 patients. Jt Bone Spine. 2014;81(5):456–8.CrossRefGoogle Scholar

Copyright information

© Japanese Society for Oral and Maxillofacial Radiology and Springer Nature Singapore Pte Ltd. 2019

Authors and Affiliations

  • Shinya Kotaki
    • 1
    Email author
  • Shoko Gamoh
    • 2
  • Hiroaki Yoshida
    • 3
  • Takaaki Noma
    • 3
  • Kaname Tsuji
    • 3
  • Hironori Akiyama
    • 1
  • Yoritaka Yotsui
    • 1
  • Tomio Iseki
    • 3
  • Kimishige Shimizutani
    • 1
  1. 1.Department of Oral RadiologyOsaka Dental University (ODU)OsakaJapan
  2. 2.Health Promotion Division, Public Health BureauOsaka City GovernmentOsakaJapan
  3. 3.First Department of Oral and Maxillofacial SurgeryOsaka Dental University (ODU)OsakaJapan

Personalised recommendations