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Commentary: What Is the Appropriate Treatment of Immunoglobulin G4-Related Vascular Lesions?

Since immunoglobulin G4-related disease (IgG4-RD) was first reported with regard to autoimmune pancreatitis, this entity has been expanded to other organs, such as the retroperitoneum, biliary tract, salivary gland, kidney, and lung. Irrespective of the organ of origin, IgG4-RD shows common clinical and pathological features. A frequent occurrence in adult male patients, IgG4-RD is characterized by elevated serum IgG4 level and steroid sensitivity. Diffuse lymphocytic IgG4-positive plasma cell infiltration, irregular fibrosis, and phlebitis are the most common pathological features. Due to severe and possibly lethal complications, it is right to say that blood vessel involvement represents a particular form of IgG4-RD. For vascular lesions in this entity, the pathological changes in large to medium size arteries occur mainly in the adventitia, but the media and intima can be involved also. The aorta is affected most often. Inflammatory changes lead to damage of the aortic wall, directly contributing to aneurysm development or dissection. Also, retroperitoneal inflammation (fibrosis) can extend into the periaortic tissues, including the adventitia, with IgG4-positive plasma cell infiltration causing chronic periaortitis. The main feature in IgG4-RD affecting the aorta is periaortitis, but true aortitis affecting the media is most likely responsible for aneurysm development. The term ‘‘IgG4-related aortitis/periaortitis’’ was advocated in the recent consensus conference on nomenclature. An arterial lesion in IgG4-RD could be associated with aneurysms or not. On the other hand, the existence of fibrotic, inflammatory, and thickened tissue around the arteries decreases the possibility of their rupture. In comparison between non-IgG4 inflammatory abdominal aortic aneurysm (IAAA) and IgG4 IAAA, Kasashima et al. reported that aneurysmal rupture in IgG4 IAAA was less frequent than that in nonIgG4 IAAA (0% vs. 30%). Actually, there are only a few cases reported in the literature of complete or contained aneurysm rupture in IgG4-RD, but only one case with ruptured aorta due to periaortitis, as Kasashima et al. report in this issue of the JEVT. These cases have reported IAAA rupture in two of them, one with dissection of the ascending aorta, one IgG4-IAAA contained rupture, and one case with ruptured visceral artery. In these patients, two of them died, including the current case by Kasashima et al. The patient reported by Qian et al. had no follow-up presented. Five of these patients underwent surgery, three of them with an open surgical repair (OSR) and two with endovascular aneurysm