Abstract：
We report a 9-year-old Japanese girl with chronic non-bacterial osteomyelitis (CNO) accompanied by recurrent erythema nodosum (EN) which was successfully treated with salazosulfapyridine (SASP). She was referred to our hospital because of recurrent erythema on her lower extremities and persistent knee and ankle arthralgia, which had been present for approximately 1 year. Although naproxen, a nonsteroidal anti-inflammatory drug, was initiated, her symptoms frequently recurred. Magnetic resonance imaging demonstrated multiple distinct high-intensity signals in the talus bones suggestive of multiple bone oedemas. Additionally, a histological examination of erythematous lesions was consistent with the histopathological findings of EN. She was diagnosed as having CNO complicated by EN, and received 250 mg/day of SASP as a second-line treatment, which showed partial response of both skin and bone lesions. Following increase in the dose of SASP to 500 mg/day resulted in complete remission of her skin and bone lesions. In conclusion, our findings suggest that SASP is effective not only for CNO bone lesions but also for EN. SASP could serve as a second-line therapeutic option at least for some cases of CNO complicated by EN refractory to nonsteroidal anti-inflammatory drugs.
摘要：
我们报告了一名9岁的日本女孩，患有慢性非细菌性骨髓炎（CNO）并伴有复发性结节性红斑（EN），该女孩已成功使用salazo磺胺吡啶（SASP）治疗。她因下肢反复出现红斑及持续性膝踝关节痛而被转诊至我们医院,已经存在了大约一年。虽然萘普生,非甾体抗炎药（NSAID），被发起，她的症状经常复发。磁共振成像显示距骨中存在多个不同的高强度信号，提示多发性骨水肿。此外,红斑性病变的组织学检查与EN的组织病理学发现一致.她被EN诊断为CNO复杂化，并接受250毫克/天的SASP作为二线治疗，显示皮肤和骨骼病变的部分反应。SASP的剂量增加至500mg/天之后，她的皮肤和骨骼病变完全缓解。总之,我们的研究结果表明，SASP不仅对CNO骨病变有效，对EN也有效。SASP至少可以作为NSAIDs难治性EN并发CNO的一些病例的二线治疗选择。
