SAPHO: has the time come for tailored therapy?

Figueiredo, Adelaide Sofia Batalha; Oliveira, Ana Luísa; Caetano, António; Moraes-Fontes, Maria Francisca

doi:10.1007/s10067-019-04675-2

Cited by 11 publications

(15 citation statements)

References 48 publications

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“…The pathogenesis of SAPHO syndrome is still poorly understood because of its enigmatic etiology. 13 , 18 Numerous experimental studies have shown that the development and progression of this entity is induced by multifarious factors of which genetics, environmental effects, and bacterial or viral pathogens might be involved. 29 To date, two major hypotheses of the pathogenesis of SAPHO syndrome have been proposed.…”

Section: Discussionmentioning

confidence: 99%

“… 7 , 11 Osseous lesions are mainly classified as necrosis, fibrosis, hyperostosis, and aberrant calcification, and these lesions always appear to be the combination of these findings. 12 , 13 The difficulty of diagnosis of this syndrome is increased in patients with only a single bone or skin symptom because symptomatic manifestations of two sites are not merged simultaneously. 10 , 14 , 15 Moreover, representative cutaneous symptoms with an average rate of 63.5% are ordinary.…”

Section: Introductionmentioning

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: Introductionmentioning

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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“…Infliximab, etanercept, and adalimumab are the most commonly used TNF-α antagonists; however, patients with SAPHO who exhibit cutaneous manifestations do not respond well to these common therapies. 1 , 16 Recently developed cytokine-directed antibodies (e.g., anakinra, ustekinumab, secukinumab, and tocilizumab) have shown promising efficacy in some case reports and case series. 16 In addition, successful treatment of refractory SAPHO syndrome was achieved in one patient by using the JAK inhibitor tofacitinib 17 and in another patient by using apremilast (a specific phosphodiesterase-4 inhibitor); 18 those findings suggest the availability of further therapeutic options for the management of SAPHO syndrome.…”

Section: Discussionmentioning

confidence: 99%

Successful treatment of synovitis, acne, pustulosis, hyperostosis, and osteitis and paradoxical skin lesions by Tripterygium wilfordii hook f: a case report

Zhang

2020

J Int Med Res

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Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare autoinflammatory disorder without standardized therapy. Anti-tumor necrosis factor (TNF)-α agents, which have been widely used in recent treatment of SAPHO syndrome, may elicit severe paradoxical psoriasiform lesions. Therefore, physicians must reverse the paradoxical skin lesions in affected patients, while improving their clinical symptoms of SAPHO syndrome. Herein, we describe a patient with SAPHO who exhibited TNF-α antagonist-induced paradoxical skin lesions and benefitted from treatment with Tripterygium wilfordii hook f (TwHF). A 58-year-old woman with SAPHO developed paradoxical psoriasiform lesions and exacerbation of primary palmoplantar pustulosis after 7 weeks of etanercept treatment. She then received TwHF treatment, which resulted in rapid and remarkable improvement in her skin lesions and osteoarticular pain. These findings suggest that TwHF might be a suitable treatment option for patients with SAPHO who exhibit TNF-α antagonist-induced paradoxical skin lesions.

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“…Synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome is a rare auto-inflammatory condition that shares musculoskeletal and cutaneous manifestations (113). Chronic recurrent multifocal osteomyelitis (CRMO) is considered the pediatric counterpart of SAPHO syndrome, arising from sterile osteomyelitis (Figures 10A-C).…”

Section: Sapho Syndrome and Crmomentioning

confidence: 99%

Axial Spondyloarthritis: Mimics and Pitfalls of Imaging Assessment

2021

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Axial spondyloarthritis (axSpA) is a chronic inflammatory disorder that predominantly involves the axial skeleton. Imaging findings of axSpA can be divided into active changes, which include bone marrow edema, synovitis, enthesitis, capsulitis, and intra-articular effusion, and structural changes, which include erosions, sclerosis, bone fatty infiltration, fat deposition in an erosion cavity, and bone bridging or ankylosis. The ability to distinguish between imaging lesions suggestive of axSpA and artifacts or lesions suggestive of other disorders is critical for the accurate diagnosis of axSpA. Diagnosis may be challenging, particularly in early-stage disease and magnetic resonance imaging (MRI) plays a key role in the detection of subtle or inflammatory changes. MRI also allows the detection of structural changes in the subchondral bone marrow that are not visible on conventional radiography and is of prognostic and monitoring value. However, bone structural changes are more accurately depicted using computed tomography. Conventional radiography, on the other hand, has limitations, but it is easily accessible and may provide insight on gross changes as well as rule out other pathological features of the axial skeleton. This review outlines the imaging evaluation of axSpA with a focus on imaging mimics and potential pitfalls when assessing the axial skeleton.

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SAPHO: has the time come for tailored therapy?

Cited by 11 publications

References 48 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

Successful treatment of synovitis, acne, pustulosis, hyperostosis, and osteitis and paradoxical skin lesions by Tripterygium wilfordii hook f: a case report

Axial Spondyloarthritis: Mimics and Pitfalls of Imaging Assessment

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