Division of Rheumatology, DuPont Children's Hospital, 1600 Rockland Road, Wilmington, DE 19899, USA.
J Rheumatol. 2012 Sep;39(9):1888-92. doi: 10.3899/jrheum.120156. Epub 2012 Aug 1.
To put forward a new concept--Blau arteritis, a form of large-vessel vasculitis phenotypically related to Takayasu disease but genetically and clinically part of an expanded phenotype of Blau syndrome.
We provide a clinical description of a new case and summarize previously published cases of arteritis associated with Blau syndrome. Genetic testing was performed by direct sequencing of exon 4 of the NOD2 gene.
The case described and those reviewed from the literature demonstrate the emerging phenotype of Takayasu-like arteritis in patients with Blau syndrome. Although most patients described to date depict an otherwise classic Blau syndrome phenotype, the current case was atypical in that the predominant features were arteritic. A novel substitution, G464W, in a highly conserved position near the nucleotide oligomerization domain of the NOD2 protein is also described.
Blau arteritis can be observed in the context of both typical and atypical (incomplete) Blau syndrome. The associated mutation in the NOD2 gene raises the question of the potential importance of this gene among patients with "primary" forms of Takayasu arteritis.
提出一个新概念——Blau 动脉炎,一种与 Takayasu 病表型相关的大血管血管炎,但在遗传和临床方面是 Blau 综合征扩展表型的一部分。
我们提供了一个新病例的临床描述,并总结了先前发表的与 Blau 综合征相关的动脉炎病例。通过直接测序 NOD2 基因的外显子 4 进行基因检测。
描述的病例和文献回顾中的病例均表明,Blau 综合征患者出现 Takayasu 样动脉炎的新兴表型。尽管迄今为止描述的大多数患者均表现为典型的 Blau 综合征表型,但当前的病例是非典型的,主要表现为动脉炎。还描述了一种位于 NOD2 蛋白核苷酸寡聚化结构域附近高度保守位置的新型取代,G464W。
Blau 动脉炎可在典型和非典型(不完全)Blau 综合征中观察到。NOD2 基因的相关突变提出了该基因在“原发性”Takayasu 动脉炎患者中的潜在重要性问题。