Takayasu arteritis associated with autoimmune/inflammatory syndrome induced by adjuvants: a case-based review

Abstract

Takayasu's arteritis (TA) is a chronic granulomatous vasculitis that predominantly affects the aorta and its major branches. Despite advancements in the understanding of the pathogenic pathways of vascular inflammation, the etiology and predisposing factors of TA remain to be fully understood. In susceptible individuals, exposure to adjuvants may trigger, unlock or unmask an autoimmune disorder, presenting as non-specific constitutional symptoms or a fully developed autoimmune syndrome such as vasculitis. Here, we hypothesize that TA could be triggered by siliconosis, a subtype of the autoimmune/inflammatory syndrome induced by adjuvants (ASIA). ASIA, also known as Shoenfeld syndrome, encompasses a wide range of autoimmune and immune-mediated diseases resulting from dysregulation of the immune response after exposure to agents with adjuvant activity. This case report describes the development of large artery vasculitis, TA, in an individual one year following the placement of silicone breast implants. The patient initially presented with non-specific symptoms, and multiple imaging methods were employed, including ultrasound diagnostics, CT angiography, and 18-fluorodeoxyglucose positron emission tomography/CT. These techniques revealed vasculitic alterations in the carotid arteries and thoracic aorta. Initial treatment with glucocorticosteroids proved ineffective, prompting the addition of steroid-sparing immunosuppressive agents. Due to the distinct clinical symptoms, disease progression, implant-associated fibrosis, and resistance to therapy, the potential involvement of implants in the development of large-vessel vasculitis was considered, and a potential association with ASIA was postulated. Although there is limited evidence to support a direct link between adjuvants and the pathogenesis of TA, similarities in cellular immunity between the two conditions exist. The diagnosis of this complex and potentially debilitating condition requires a comprehensive clinical examination, laboratory evaluation, and instrumental assessment. This will aid in identifying potential contributing factors and ensuring successful treatment.

Keywords: Autoimmune/inflammatory syndrome induced by adjuvants; Autoimmunity; Breast implants; Case report; Immunologic adjuvants; Large vessel vasculitis; Silicone gels; Takayasu arteritis.

Fig. 1

Images of 18-FDG PET/CT and three-dimensional computed tomography showing: A an 18-FDG PET/CT showing an increased metabolic activity in the wall of the ascending aorta and aortic arch. Increased glucose metabolism is also detected around the implants and paraspinal muscles suggesting pericapsular inflammation and paraspinal myositis. B An 18-FDG PET/CT showing heightened glucose metabolism in lower legs and upper extremity proximal muscles. C A three-dimensional computed tomography (3D CT) reconstruction image of the descending aorta stenosis

Fig. 2

Differences in the angiographic findings of the thoracic aorta between 2020 (A) and 2022 (B) show a significant regression in the thickness of the aortic wall and an increase in the vascular lumen

Fig. 3

Flow diagram showing the number of identified, screened, eligible and included studies