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伴有延迟性皮肤表现的布劳综合征:一例报告

Blau Syndrome With Delayed Cutaneous Manifestations: A Case Report.

作者信息

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

机构信息

Department of Pathology, Loyola University Medical Center, Maywood, IL; and.

Department of Dermatology, Loyola University Medical Center, Maywood, IL.

出版信息

Am J Dermatopathol. 2024 Jun 1;46(6):381-382. doi: 10.1097/DAD.0000000000002715. Epub 2024 Apr 23.

DOI:10.1097/DAD.0000000000002715
PMID:38648024
Abstract

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.

摘要

布劳综合征是一种罕见的家族性自身炎症性疾病,其特征为肉芽肿性皮炎、多关节炎和葡萄膜炎三联征。布劳综合征呈现常染色体显性遗传模式,可由核苷酸结合寡聚化结构域2(NOD2)功能获得性突变引起,NOD2是模式识别受体的NOD样受体家族成员。NOD2突变导致炎性细胞因子上调并引发自身炎症。由于该病症罕见且症状出现早,布劳综合征可能被误诊为青少年特发性关节炎。我们报告一例37岁男性患者,有长期记录的青少年特发性关节炎和葡萄膜炎病史,其躯干和上肢出现无症状的粉红色丘疹疹。活检显示为非干酪样、形态良好的真皮肉芽肿,淋巴细胞炎症相对稀疏,并有朗格汉斯型巨细胞。基因检测证实NOD2存在突变。根据患者的临床病史、组织学发现、基因检测结果,作出了布劳综合征的诊断。

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引用本文的文献

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Blau Syndrome (Juvenile Systemic Granulomatosis): State-Of-The-Art Review.布劳综合征(青少年系统性肉芽肿病):最新综述
Pediatr Dermatol. 2025 Jul-Aug;42(4):728-734. doi: 10.1111/pde.16001. Epub 2025 Jun 2.