Case report

Yang, Qiao; Zhao, Yixia; Li, Chen; Luo, Yaping; Hao, Weixin; Zhang, Wen

doi:10.1097/md.0000000000011149

Cited by 43 publications

(10 citation statements)

References 15 publications

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“…Lastly, JAKi could improve autoinflammatory disorders. A patient with synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO), has been successfully treated by a combination of methotrexate and oral tofacitinib (200). Similarly, the clinical trials NCT01724580 and NCT02974595 proved efficacy of baricitinib in a small group of patients affected by CANDLE (chronic atypical neutrophilic dermatosis with lipodystrophy and elevated temperatures) and SAVI (stimulator of IFN-genes associated vasculopaty with onset in infancy) (201, 202).…”

Section: Possible Future Applicationsmentioning

confidence: 99%

Emerging Topical and Systemic JAK Inhibitors in Dermatology

2019

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Accumulating data on cellular and molecular pathways help to develop novel therapeutic strategies in skin inflammation and autoimmunity. Examples are psoriasis and atopic dermatitis, two clinically and immunologically well-defined disorders. Here, the elucidation of key pathogenic factors such as IL-17A/IL-23 on the one hand and IL-4/IL-13 on the other hand profoundly changed our therapeutic practice. The knowledge on intracellular pathways and governing factors is shifting our attention to new druggable molecules. Multiple cytokine receptors signal through Janus kinases (JAKs) and associated signal transducer and activators of transcription (STATs). Inhibition of JAKs can simultaneously block the function of multiple cytokines. Therefore, JAK inhibitors (JAKi) are emerging as a new class of drugs, which in dermatology can either be used systemically as oral drugs or locally in topical formulations. Inhibition of JAKs has been shown to be effective in various skin disorders. The first oral JAKi have been recently approved for the treatment of rheumatoid arthritis and psoriatic arthritis. Currently, multiple inhibitors of the JAK/STAT pathway are being investigated for skin diseases like alopecia areata, atopic dermatitis, dermatomyositis, graft-versus-host-disease, hidradenitis suppurativa, lichen planus, lupus erythematosus, psoriasis, and vitiligo. Here, we aim to discuss the immunological basis and current stage of development of JAKi in dermatology.

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Section: Possible Future Applicationsmentioning

confidence: 99%

Emerging Topical and Systemic JAK Inhibitors in Dermatology

2019

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“…Thus, the use of IL-17-blockade may be another therapeutic option in otherwise refractory cases [ 75 ]. Individual case reports exist on successful treatment of SAPHO with apremilast, a PDE4-inhibitor [ 78 ], or the JAK inhibitor tofacitinib [ 79 , 80 ].…”

Section: Treatment Monitoring and Treat-to-target Protocolsmentioning

confidence: 99%

New Insights into Adult and Paediatric Chronic Non-bacterial Osteomyelitis CNO

Hedrich

Morbach

Reiser³

et al. 2020

Curr Rheumatol Rep

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Purpose of Review To describe in detail the clinical synopsis and pathophysiology of chronic non-bacterial osteomyelitis and SAPHO syndrome. Recent Findings Chronic non-bacterial osteomyelitis (CNO) has been identified as a disease entity for almost 50 years. This inflammatory bone disorder is characterized by osteolytic as well as hyperostotic/osteosclerotic lesions. It is chronic in nature, but it can present with episodic flairs and phases of remission, which have led to the denomination “chronic recurrent osteomyelitis”, with its severe multifocal form “chronic recurrent multifocal osteomyelitis” (CRMO). For almost three decades, an infectious aetiology had been considered, since especially Propionibacterium acnes had been isolated from bone lesions of individual patients. However, this concept has been challenged since long-term antibiotic therapy did not alter the course of disease and modern microbiological techniques (including PCR) failed to confirm bone infection as an underlying cause. Over recent years, a profound dysregulation of cytokine expression profiles has been demonstrated in innate immune cells of CNO patients. A hallmark of monocytes from CNO patients is the failure to produce immune regulatory cytokines interleukin-10 (IL-10) and IL-19, which have been linked with genetic and epigenetic alterations. Subsequently, a significant upregulation of pro-inflammatory, NLRP3 inflammasome-dependent cytokines (IL-1β and TNF-α), has been demonstrated. Summary The current knowledge on CNO, the underlying molecular pathophysiology, and modern imaging strategies are summarized; differential diagnoses, treatment options, outcome measures, as well as quality of life studies are discussed.

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“… 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. In 2017, we described a patient with SAPHO who achieved a remarkable degree of remission following TwHF treatment.…”

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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Case report

Cited by 43 publications

References 15 publications

Emerging Topical and Systemic JAK Inhibitors in Dermatology

Emerging Topical and Systemic JAK Inhibitors in Dermatology

New Insights into Adult and Paediatric Chronic Non-bacterial Osteomyelitis CNO

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

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