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 the...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.展开更多
Introduction: The localization of bone tuberculosis at the level of the olecranon is rare and can pose a problem of differential diagnosis with a tumoral affection. Only the biopsy allows the diagnosis of certainty. T...Introduction: The localization of bone tuberculosis at the level of the olecranon is rare and can pose a problem of differential diagnosis with a tumoral affection. Only the biopsy allows the diagnosis of certainty. The goal was to report our first case to do a review of the literature. Clinical Observation: This was a 64-year-old patient who consulted 2 years after the onset of symptoms for swelling and mechanical pain in the left elbow. On clinical examination there was a firm consistent mass measuring 6/4 cm in diameter, not very painful and hot on the posterior surface of the left elbow, accompanied by paresthesias in the territory of the ulnar nerve associated with partial functional impotence of the left forearm with little altered general condition. The X-rays of the left elbow showed extensive bone lysis of the olecranon with fracture of the base of the olecranon. The CT Scan of the elbow performed showed osteolysis of the olecranon with extensive bone reactions at the distal end of the humerus. The biopsy carried out with histological examination concluded with an aspect of tuberculous osteitis and the culture came back sterile. A curettage was performed associated with anti-tuberculosis treatment for 12 months as well as the placement of a posterior splint. The evolution at 9 months was unsatisfactory with persistence of paresis and stiffness of the elbow. Conclusion: Tuberculous osteitis of the olecranon can simulate a malignant tumor with non-specific signs of bone tuberculosis. Only the anatomopathological examination is definitive for the diagnosis. The treatment is multidisciplinary. Orthopedic evolution is difficult to predict.展开更多
文摘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.
文摘Introduction: The localization of bone tuberculosis at the level of the olecranon is rare and can pose a problem of differential diagnosis with a tumoral affection. Only the biopsy allows the diagnosis of certainty. The goal was to report our first case to do a review of the literature. Clinical Observation: This was a 64-year-old patient who consulted 2 years after the onset of symptoms for swelling and mechanical pain in the left elbow. On clinical examination there was a firm consistent mass measuring 6/4 cm in diameter, not very painful and hot on the posterior surface of the left elbow, accompanied by paresthesias in the territory of the ulnar nerve associated with partial functional impotence of the left forearm with little altered general condition. The X-rays of the left elbow showed extensive bone lysis of the olecranon with fracture of the base of the olecranon. The CT Scan of the elbow performed showed osteolysis of the olecranon with extensive bone reactions at the distal end of the humerus. The biopsy carried out with histological examination concluded with an aspect of tuberculous osteitis and the culture came back sterile. A curettage was performed associated with anti-tuberculosis treatment for 12 months as well as the placement of a posterior splint. The evolution at 9 months was unsatisfactory with persistence of paresis and stiffness of the elbow. Conclusion: Tuberculous osteitis of the olecranon can simulate a malignant tumor with non-specific signs of bone tuberculosis. Only the anatomopathological examination is definitive for the diagnosis. The treatment is multidisciplinary. Orthopedic evolution is difficult to predict.
