伴有延迟性皮肤表现的布劳综合征:病例报告

Elnaz Panah, Erin Garfield, Zisansha Zahirsha, Aaron Muhlbauer, Eden Lake, Jodi Speiser
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摘要

布劳综合征是一种罕见的家族性自身炎症性疾病,以肉芽肿性皮炎、多关节炎和葡萄膜炎三联症为特征。布劳综合征为常染色体显性遗传,可由核苷酸结合寡聚化结构域 2(NOD2)的功能增益突变引起,NOD2 是模式识别受体 NOD 样受体家族的成员。NOD2 基因突变会导致炎症细胞因子上调,从而引发自身炎症。由于该病罕见且发病较早,布劳综合征可能会被误诊为幼年特发性关节炎。我们介绍了一例 37 岁的男性患者,他有长期的幼年特发性关节炎和葡萄膜炎病史,躯干和上肢出现无症状的粉红色丘疹。活组织检查显示,患者的真皮肉芽肿为非溃疡性、形态良好的肉芽肿,伴有相对稀疏的淋巴细胞炎症和朗格汉斯型巨细胞。基因检测证实了 NOD2 基因突变。根据患者的临床病史、组织学检查结果和基因检测结果,诊断为布劳综合征。
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Blau Syndrome With Delayed Cutaneous Manifestations: A Case Report.
Blau syndrome is a rare familial autoinflammatory disorder characterized by the triad of granulomatous dermatitis, polyarthritis, and uveitis. Blau syndrome exhibits an autosomal dominant inheritance pattern and can be caused by a gain-of-function mutation in nucleotide-binding oligomerization domain 2 (NOD2), a member of the NOD-like receptor family of pattern recognition receptors. Mutations in NOD2 cause upregulation of inflammatory cytokines and resultant autoinflammation. Because of the rarity of this condition and early onset of symptoms, Blau syndrome may be misdiagnosed as juvenile idiopathic arthritis. We present a case of a 37-year-old male patient with a long-documented history of juvenile idiopathic arthritis and uveitis, who developed an asymptomatic eruption of pink papules on the trunk and upper extremities. A biopsy demonstrated noncaseating, well-formed dermal granulomas with relatively sparse lymphocytic inflammation and Langerhans-type giant cells. Genetic testing confirmed a mutation in NOD2. Based on the patient's clinical history, histologic findings, genetic testing, the diagnosis of Blau syndrome was made.
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