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

Kotaki, Shinya; Gamoh, Shoko; Yoshida, Hiroaki; Noma, Takaaki; Tsuji, Kaname; Akiyama, Hironori; Yotsui, Yoritaka; Iseki, Tomio; Shimizutani, Kimishige

doi:10.1007/s11282-019-00405-1

Cited by 7 publications

(5 citation statements)

References 12 publications

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“…Involvement of the femur in SAPHO syndrome comprises various bone lesions, such as hyperostosis, osteomyelitis, bone marrow fibrosis, osteonecrosis, osteolysis, mineralization, osteosclerosis, and bone resorption and formation. 14 , 15 , 24 In fact, osseous lesions emerging from the femur are a mixture of the above-mentioned factors. In our case, osteomyelitis and cortical sclerosis were found in the right proximal femoral diaphysis.…”

Section: Discussionmentioning

confidence: 99%

“…7 Osseous lesions in SAPHO syndrome are commonly found in the anterior chest wall, especially in the sternoclavicular and sternocostal joints. 15,24,25 Spinal complaints are the second most common symptom in SAPHO syndrome and lesions are located in axial bone sites. 26 However, peripheral long bone involvement, such as the femur, in SAPHO syndrome is rare.…”

Section: Discussionmentioning

confidence: 99%

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Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome of femoral neoplasm-like onset: a case-based review

et al. 2021

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Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is an umbrella term covering a constellation of bone lesions and skin manifestations, but has rarely been clarified in the clinic. We report a 28-year-old woman who had initial onset of SAPHO syndrome with involvement of the femur, and she experienced a tortuous diagnostic course. We also performed a literature review of SAPHO syndrome cases involving the femur and summarize several empirical conclusions by integrating previous findings with our case. Furthermore, we propose our perspective that ailment of the skin caused by infection of pathogens might be the first hit for triggering or perpetuating the activation of the immune system. As a result, musculoskeletal manifestations are probably the second hit by crosstalk of an autoimmune reaction. The skin manifestations preceding bone lesions can be well explained. Current interventions for SAPHO syndrome remain controversial, but drugs aiming at symptom relief could serve as the first preference for treatment. An accurate diagnosis and appropriate treatment can cure patients in a timely manner. Although the pathogenesis of SAPHO syndrome remains to be determined, physicians and surgeons still need to heighten awareness of this entity to avoid invasive procedures, such as frequent biopsies or nonessential ostectomy.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome of femoral neoplasm-like onset: a case-based review

et al. 2021

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“…The main characteristics of the dermatologic manifestations are neutrophilic pustular dermatoses, 24 including pustular psoriasis, palm plant impetigo (PPP), psoriasis vulgaris, severe acne, or hidradenitis suppurativa 9,25,26 . Chronic inflammatory aseptic osteitis is the central feature of SAPHO bone lesions, manifested by tenderness, pain, and changes in bone structure 7,27 . Moreover, the routine blood test showed that the inflammatory markers hs‐CRP and ESR were significantly increased in SAPHO syndrome samples 28 .…”

Section: Discussionmentioning

confidence: 99%

“…9, 25,26 Chronic inflammatory aseptic osteitis is the central feature of SAPHO bone lesions, manifested by tenderness, pain, and changes in bone structure. 7,27 Moreover, the routine blood test showed that the inflammatory markers hs-CRP and ESR were significantly increased in SAPHO syndrome samples. 28 Meanwhile, increased Th17 cells can be determined in the peripheral blood of SAPHO patients, 29 which help neutrophils tend to the site of inflammation.…”

Section: Discussionmentioning

confidence: 99%

An integrated transcriptomics and network pharmacology approach to explore the mechanism of Wang‐Bi tablet against SAPHO syndrome

Wang,

Gong,

Xia

et al. 2024

Int J of Rheum Dis

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BackgroundSAPHO syndrome is recognized as a rare entity with damage to skin and bones due to inflammation. Currently, the treatment for SAPHO syndrome is still a challenge in clinical practice. In this study, an integrated transcriptomics and network pharmacology approach was applied to explore the therapeutic effect and mechanism of Wang‐Bi tablet (WBT) on SAPHO syndrome.MethodsThe main components of WBT and their targets, as well as the targets of SAPHO syndrome, were collected from databases. Network visualization was performed using Cytoscape software. The GO and KEGG enrichment analysis was executed by David dataset. Then, the molecular mechanism of WBT improving SAPHO syndrome was validated by transcriptomics of peripheral blood neutrophils in SAPHO syndrome. Finally, the above results were validated by molecular docking.ResultsThe Network Pharmacology results showed there are 152 core targets for WBT treatment on SAPHO syndrome. RNA‐seq data showed 442 differentially expressed genes (DEGs) in peripheral blood neutrophils of SAPHO patients. Intriguingly, NIK/NF‐kappaB‐, MyD88‐dependent toll‐like receptor‐, and MAPK pathway were included in the enrichment results of network pharmacology and RNA‐seq. Moreover, we verified that the core components of WBT have good affinity with the core targets of NIK/NF‐kappaB‐, MyD88‐dependent toll‐like receptor‐, and MAPK pathway by molecular docking.ConclusionsThis study illustrated that the possible mechanisms of WBT against SAPHO syndrome may be related to NIK/NF‐kappaB‐, MyD88‐dependent toll‐like receptor‐, and MAPK pathway, and further experiments are needed to prove these predictions.

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“…According to a study conducted by Ji Rak Kim et al [ 5 ], a small percentage of subjects (15%) exhibited ANA/RF positivity. Furthermore, Shinya Kototaki et al [ 6 ] reported a high prevalence of severe TMD in patients with SAPHO syndrome. According to Alina Grozdinska et al [ 7 ], there appears to be a statistical relationship between TMD and Hashimoto’s thyroiditis (HT).…”

Section: Introductionmentioning

confidence: 99%

Etiologic Factors of Temporomandibular Disorders: A Systematic Review of Literature Containing Diagnostic Criteria for Temporomandibular Disorders (DC/TMD) and Research Diagnostic Criteria for Temporomandibular Disorders (RDC/TMD) from 2018 to 2022

Warzocha,

Gadomska-Krasny,

Mrowiec

2024

Healthcare

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This study aims to conduct a systematic analysis of literature published between 1 January 2018 and 1 September 2022, exploring factors influencing the progression or development of temporomandibular disorders (TMD), diagnosed using the Diagnostic Criteria for Temporomandibular Disorders (DC/TMD) or Research Diagnostic Criteria for Temporomandibular Disorders (RDC/TMD). Three electronic databases were reviewed to identify papers that examined TMD factors using DC/TMD or RDC/TMD. Inclusion criteria encompassed original research published in English between 1 January 2018 and 1 October 2022, online, and complete DC/TMD or RDC/TMD studies on human participants aged 18 or older. Two authors independently assessed the risk of bias using The Joanna Briggs Institute (JBI) Analytical cross-sectional studies’ Critical Appraisal Tool. Of 1478 articles, 11 were included. The studies revealed strong associations between TMD and factors such as female, poor sleep quality, depression, oral parafunction, anxiety, somatization, and anatomical features. However, variables such as education, living conditions, socioeconomic status, marital status, chronic pain, and stress did not exhibit statistically significant correlations. Based on the obtained data, it can be concluded that the causes of TMD are largely related to psychological factors, which supports the biopsychosocial theory of the disorder.

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

Cited by 7 publications

References 12 publications

Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome of femoral neoplasm-like onset: a case-based review

Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome of femoral neoplasm-like onset: a case-based review

An integrated transcriptomics and network pharmacology approach to explore the mechanism of Wang‐Bi tablet against SAPHO syndrome

Etiologic Factors of Temporomandibular Disorders: A Systematic Review of Literature Containing Diagnostic Criteria for Temporomandibular Disorders (DC/TMD) and Research Diagnostic Criteria for Temporomandibular Disorders (RDC/TMD) from 2018 to 2022

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