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[将儿童结节病误诊为非朗格汉斯细胞组织细胞增多症并使用肿瘤坏死因子-α拮抗剂治疗]

[Misdiagnosed childhood sarcoidosis as non-Langerhans' cell histiocytosis treated with tumor necrosis factors-α antagonists].

作者信息

Petiti Martin G, Castellanos González M, Sanz Bueno J, Burgués Calderón M, Villar Buil M, Vanaclocha F, Peralto J L

机构信息

Servicio de Dermatología y Anatomía Patológica, Hospital Universitario 12 de Octubre, Madrid, España.

出版信息

An Pediatr (Barc). 2012 Oct;77(4):267-71. doi: 10.1016/j.anpedi.2012.01.024. Epub 2012 May 17.

DOI:10.1016/j.anpedi.2012.01.024
PMID:22608978
Abstract

Sarcoidosis is a chronic multisystemic granulomatous disease of unknown origin. Generalised eruptive histiocytosis is a rare, benign, self-healing, non-Langerhans' cell histiocytosis (non-LCH). We report the case of an 8-year-old girl with sarcoidosis who was misdiagnosed as non-LCH. She was treated with oral corticosteroids, methotrexate and adalimumab, but there was insufficient control of ocular disease. The introduction of infliximab achieved a control of the uveitis and enabled the corticosteroid dose to be tapered. In some cases of sarcoidosis the lack of well-organised granuloma formation at the beginning of the disease, and the presence of prominent giant cells may suggest alternative diagnoses, such as non-LCH. Although the experience of tumour necrosis factor-α antagonists use in children with sarcoidosis is limited, these drugs may be helpful for those patients experiencing a severe and refractory disease.

摘要

结节病是一种病因不明的慢性多系统肉芽肿性疾病。全身性发疹性组织细胞增多症是一种罕见的、良性的、可自愈的非朗格汉斯细胞组织细胞增多症(非LCH)。我们报告了一例8岁结节病女孩被误诊为非LCH的病例。她接受了口服糖皮质激素、甲氨蝶呤和阿达木单抗治疗,但眼部疾病控制不佳。英夫利昔单抗的使用实现了葡萄膜炎的控制,并使糖皮质激素剂量得以逐渐减少。在某些结节病病例中,疾病初期缺乏组织良好的肉芽肿形成以及存在突出的巨细胞可能提示其他诊断,如非LCH。尽管肿瘤坏死因子-α拮抗剂在儿童结节病中的使用经验有限,但这些药物可能对那些患有严重难治性疾病的患者有帮助。

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[Misdiagnosed childhood sarcoidosis as non-Langerhans' cell histiocytosis treated with tumor necrosis factors-α antagonists].[将儿童结节病误诊为非朗格汉斯细胞组织细胞增多症并使用肿瘤坏死因子-α拮抗剂治疗]
An Pediatr (Barc). 2012 Oct;77(4):267-71. doi: 10.1016/j.anpedi.2012.01.024. Epub 2012 May 17.
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[Clarifications on the article "sarcoidosis simulating a non-Langerhans cell histiocytosis, treated with tumour necrosis factor antagonists"].[关于“用肿瘤坏死因子拮抗剂治疗的类似非朗格汉斯细胞组织细胞增多症的结节病”一文的澄清]
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Refractory chronic cutaneous sarcoidosis responsive to dose escalation of TNF-alpha antagonists.对肿瘤坏死因子-α拮抗剂剂量递增有反应的难治性慢性皮肤结节病
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Cutaneous sarcoidosis occurring during anti-TNF-alpha treatment: report of two cases.在抗 TNF-α 治疗期间发生的皮肤结节病:两例报告。
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Adalimumab successful in sarcoidosis patients with refractory chronic non-infectious uveitis.阿达木单抗治疗难治性慢性非感染性葡萄膜炎的疗效。
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Infliximab and adalimumab for uveitis.英夫利昔单抗和阿达木单抗治疗葡萄膜炎。
Ocul Immunol Inflamm. 2012 Feb;20(1):18-26. doi: 10.3109/09273948.2011.633205.

引用本文的文献

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The Efficacy and Safety of Infliximab in Refractory Noninfectious Uveitis: A Meta-Analysis of Observational Studies.英夫利昔单抗治疗难治性非感染性葡萄膜炎的疗效和安全性:观察性研究的荟萃分析
Front Pharmacol. 2021 Sep 16;12:620340. doi: 10.3389/fphar.2021.620340. eCollection 2021.