Rapid remission of refractory synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome in response to the Janus kinase inhibitor tofacitinib: A case report

Abstract

BACKGROUND Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare autoinflammatory disease for which clinical treatment has not been standardized. Janus kinase (JAK) inhibitors represent a novel therapeutic option for rheumatoid arthritis, psoriatic arthritis, and some other autoinflammatory diseases. However, the clinical utility of JAK inhibitors in treating SAPHO syndrome has not been thoroughly investigated. In this study, we describe a patient with SAPHO syndrome who failed to respond to conventional treatment but demonstrated a remarkable and rapid response to the JAK inhibitor tofacitinib. CASE SUMMARY A 62-year-old female patient presented with swelling and pain at the sternoclavicular joints, back pain that limited her activities, arthralgia in the right knee, and cutaneous lesions. Her symptoms were unresponsive to nonsteroidal anti-inflammatory drugs, disease-modifying antirheumatic drugs, Tripterygium wilfordii hook f, and bisphosphonates. SAPHO syndrome was diagnosed in accordance with dermatological and osteoarticular manifestations and abnormal inflammatory factors. Multiple image studies have illustrated bone lesions and pathological fractures of vertebral bodies. Oral treatment with tofacitinib at 5 mg twice daily with methotrexate and bisphosphonates was initiated. The patient reported that her pain symptoms were relieved after 3 d and her cutaneous lesions were reduced after 4 wk of treatment. Vertebral lesions were improved after 6 mo on tofacitinib. No serious adverse effects were noted. CONCLUSION JAK inhibitor therapy may be a promising strategy to treat SAPHO syndrome.

Tofacitinib for the treatment of erythematotelangiectatic and papulopustular rosacea: A retrospective case series

“…Tofacitinib used in our study is a JAK1/3 inhibitor, approved for the treatment of psoriatic arthritis, rheumatoid arthritis and ulcerative colitis 10 . Furthermore, JAK1/3 inhibitors have achieved satisfactory therapeutic effects in the treatment of a variety of inflammatory skin diseases, including alopecia areata, vitiligo, psoriasis, systemic lupus erythematosus, atopic dermatitis and hidradenitis suppurativa 11–14 . For instance, A meta‐analysis showed that 57.8% of vitiligo patients had a good response or repigmentation rate (>50%) when receiving JAK inhibitors including tofacitinib and ruxolitinib.…”

“…10 Furthermore, JAK1/3 inhibitors have achieved satisfactory therapeutic effects in the treatment of a variety of inflammatory skin diseases, including alopecia areata, vitiligo, psoriasis, systemic lupus erythematosus, atopic dermatitis and hidradenitis suppurativa. [11][12][13][14] For instance, A meta-analysis showed that 57.8% of vitiligo patients had a good response or repigmentation rate (>50%) when receiving JAK inhibitors including tofacitinib and ruxolitinib. The favorable response rate increased to 88.9% in patients receiving both a JAK inhibitor and phototherapy.…”

A Case of SAPHO Syndrome Complicated by Uveitis with Good Response to Both TNF Inhibitor and JAKinib

“…A 44-year-old woman with SAPHO was treated with tofacitinib 5 mg BD dose with marked improvement in arthralgia by 4 weeks [ 22 ]. Another case report of a 62-year-old female had significant improvement in cutaneous and articular manifestations after 4 weeks of tofacitinib [ 23 ]. Our patient had a good cutaneous and articular response after 1 month of tofacitinib use but could not afford the medicine due to financial constraints.…”

New Insights in the Treatment of SAPHO Syndrome and Medication Recommendations

“…Secukinumab (Cosentyx ® ) is a first-in-class fully human monoclonal antibody against interleukin-17A. 69 After receiving secukinumab, 87.5% (7/8) of patients with cutaneous symptoms achieved satisfactory results; however, only 66.7% (6/9) with osteoarticular symptoms improved ( Table 1 ). Cornillier et al 62 reported a 44-year-old patient suffering from SAPHO syndrome for ten years.…”

“…Liu et al 68 presented a patient with SAPHO syndrome complicated by lymphangioleiomyomatosis whose arthralgia and pulmonary function improved after tofacitinib treatment. Li et al 69 reported a 62-year-old female patient presenting with swelling and pain at the sternoclavicular joints, back pain that limited her activities, arthralgia in the right knee, and cutaneous lesions. A combination oral treatment with tofacitinib 5 mg twice daily with methotrexate and bisphosphonates was administered.…”