Park Su-Kyung, Hwang Pyung-Han, Yun Seok-Kweon, Kim Han-Uk, Park Jin
Department of Dermatology, Chonbuk National University Medical School, Jeonju, Korea.
Department of Pediatrics, Chonbuk National University Medical School, Jeonju, Korea.; Research Institute of Clinical Medicine of Chonbuk National University-Biomedical Research Institute of Chonbuk National University Hospital, Jeonju, Korea.
Ann Dermatol. 2017 Feb;29(1):74-78. doi: 10.5021/ad.2017.29.1.74. Epub 2017 Feb 3.
The development of cutaneous sarcoidosis as a paradoxical adverse event of tumor necrosis factor alpha (TNF-α) blockers has been reported in the literature; however, an erythrodermic form of cutaneous sarcoidosis during anti-TNF-α therapy has not yet been reported. Herein, we report the first case of an erythrodermic form of cutaneous sarcoidosis during anti-TNF-α therapy and review previous studies of cutaneous sarcoidosis. A 6-year-old Korean girl who had been suffering from juvenile rheumatoid arthritis presented with generalized erythematous skin eruption involving more than about 90% of her body surface area. After 14 months of etanercept treatment, the new erythematous skin eruption had developed and progressed into generalized erythroderma. Exclusion of suspected co-medication had been performed based on medication history. She had no other systemic symptoms, and ophthalmologic and neurologic examinations were normal. Histopathologic findings of the skin lesion revealed diffuse non-caseating granulomatous infiltrates composed of epithelioid histiocytes with sparse lymphocytes involving the entire dermis. Periodic-acid-Schiff and acid-fast stains were negative, and acid-fast bacilli was not detected by polymerase chain reaction of the skin biopsy. Based on clinicopathologic findings, she was diagnosed with etanercept-induced sarcoidal granuloma. After discontinuation of the suspected agent, the lesions spontaneously disappeared.
文献中已报道皮肤结节病作为肿瘤坏死因子α(TNF-α)阻滞剂的一种矛盾性不良事件的发生情况;然而,抗TNF-α治疗期间出现红皮病型皮肤结节病的情况尚未见报道。在此,我们报告首例抗TNF-α治疗期间发生的红皮病型皮肤结节病病例,并回顾以往关于皮肤结节病的研究。一名6岁韩国女童,患有幼年类风湿关节炎,全身出现红斑性皮肤疹,累及身体表面积约90%以上。接受依那西普治疗14个月后,新的红斑性皮肤疹出现并发展为全身性红皮病。根据用药史排除了可疑的合并用药。她无其他全身症状,眼科和神经科检查均正常。皮肤病变的组织病理学检查发现,整个真皮层有由上皮样组织细胞组成的弥漫性非干酪样肉芽肿浸润,淋巴细胞稀少。过碘酸-希夫染色和抗酸染色均为阴性,皮肤活检的聚合酶链反应未检测到抗酸杆菌。根据临床病理检查结果,她被诊断为依那西普诱导的结节样肉芽肿。停用可疑药物后,皮损自行消退。