Simultaneous development of sarcoidosis and cutaneous vasculitis in a patient with refractory Crohn's disease during infliximab therapy.
Simultaneous development of sarcoidosis and cutaneous vasculitis in a patient with refractory Crohn's disease during infliximab therapy.
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DOI：
10.1186/s12890-016-0193-5
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发表时间：
2016-02-11
影响因子：
3.1
通讯作者：
Ichinose M
中科院分区：
文献类型：
--
作者：
Numakura T;Tamada T;Nara M;Muramatsu S;Murakami K;Kikuchi T;Kobayashi M;Muroi M;Okazaki T;Takagi S;Eishi Y;Ichinose M
抗TNF-α治疗期间的副作用炎症定义为免疫反应而非感染因子诱导的副作用，如银屑病样皮肤病变、葡萄膜炎和结节病样肉芽肿。在这里，我们报告了一个非常罕见的情况下，同时发展结节病和皮肤血管炎的难治性克罗恩病患者在英夫利西单抗治疗期间，这两个自发解决没有停止英夫利西单抗。2000年9月，一名23岁的日本男性被诊断出患有克罗恩病。泼尼松龙与美沙拉秦的联合用药最初被引入，并成功了近一年。2002年6月，由于他的胃肠道症状复发且难治性，引入英夫利昔单抗（IFX）5 mg/kg治疗。2011年2月，由于他几乎每次静脉注射IFX都反复出现关节痛，在诊断为IFX继发性失败的情况下，将IFX剂量增加至10 mg/kg。2012年12月，患者诉轻微干咳和双下肢皮疹瘙痒，因胸部X线检查显示双侧肺门淋巴结肿大而转诊至我院。胸部电脑断层显示双侧肺门淋巴结肿大，双侧上肺有细小网状结节影。血清钙、血管紧张素转换酶和可溶性白细胞介素2受体水平未升高，但抗核抗体滴度显著升高。仔细排除分枝杆菌感染。经支气管肺活检显示非干酪化上皮样细胞肉芽肿符合结节病。右肢皮肤活检诊断为白细胞破碎性血管炎。患者在抗TNF-α治疗期间被诊断为一系列反常炎症。由于他的反常炎症不严重，排除了机会性感染，IFX谨慎地继续治疗难治性克罗恩病。9个月后，不仅他的胸内病变，而且他的皮肤病变也自发消退。治疗抗TNF-α治疗患者的医生应了解，在仔细排除感染因子和对患者可能的风险和获益进行仔细评估的基础上，矛盾炎症可以在不停止抗TNF-α治疗的情况下得到解决。
Paradoxical inflammations during anti-TNF-α therapy are defined as adverse effects such as psoriasiform skin lesions, uveitis and sarcoidosis-like granulomas induced by immune reactions, not by infectious agents. Here, we report a very rare case of the simultaneous development of sarcoidosis and cutaneous vasculitis in a patient with refractory Crohn’s disease during infliximab therapy and both of which resolved spontaneously without the cessation of infliximab. In September 2000, 23-year old Japanese male was diagnosed with Crohn’s disease. Prednisolone in combination with mesalazine was introduced at first and succeeded for almost one year. In June 2002, since his gastrointestinal symptoms relapsed and were refractory, infliximab (IFX) therapy 5 mg/kg was introduced. In February 2011, because he had repeated arthralgia almost every intravenous IFX administration, IFX was increased to 10 mg/kg under the diagnosis of a secondary failure of IFX. In December 2012, he complained of slight dry cough and an itchy eruption on both lower limbs, and he was referred to our hospital due to the appearance of bilateral hilar lymphadenopathy on chest X-ray examination. Chest computed tomogram revealed bilateral hilar lymphadenopathy and fine reticulonodular shadows on the bilateral upper lungs. Serum calcium, angiotensin-converting enzyme and soluble interleukin 2 receptor levels were not elevated, but the titer of antinuclear antibody was considerably elevated. Mycobacterium infection was carefully excluded. Trans-bronchial lung biopsy showed non-caseating epithelioid cell granulomas compatible with sarcoidosis. The skin biopsy of the right limb was diagnosed as leukocytoclastic vasculitis. The patient was diagnosed as having a series of paradoxical inflammations during anti-TNF-α therapy. Since his paradoxical inflammations were not severe and opportunistic infections were excluded, IFX was cautiously continued for refractory Crohn’s disease. Nine months later, not only his intrathoracic lesions but also his cutaneous lesions had spontaneously resolved. Physicians caring for patients with anti-TNF-α therapy should know that, based on a careful exclusion of infectious agents and thoughtful assessment of the patient’s possible risks and benefits, paradoxical inflammations can be resolved without the cessation of anti-TNF-α therapy.