Three Case Reports of Adult-onset Still Disease Associated with Breast Implantation

Author:

Maitani Kosuke1,Kadowaki Miki1,Yamagiwa Yuko1,Yasuhara Yumiko2,Kozuki Shino1,Otsuka Yohei3,Kubo Tateki4,Tomita Koichi5

Affiliation:

1. Department of Plastic and Reconstructive Surgery, Sakai City Medical Center, Osaka, Japan

2. Department of Diagnostic Pathology, Sakai City Medical Center, Osaka, Japan

3. Department of Internal Medicine, Sakai City Medical Center, Osaka, Japan

4. Department of Plastic and Reconstructive Surgery, Osaka University Graduate School of Medicine, Osaka, Japan

5. Department of Plastic and Reconstructive Surgery, Kindai University Faculty of Medicine, Osaka, Japan.

Abstract

Summary: Autoimmune syndrome induced by adjuvants (ASIA) is an uncommon clinical condition reported by Shoenfeld et al. Although this syndrome is not scientifically validated, numerous reports on it have been published, and the manifestations are postulated to be diverse, including generalized symptoms such as chronic fatigue, myalgia, arthralgia, or dry mouth, induced by exogenous substances, specifically adjuvants, which can encompass vaccines, organisms, and silicone. Concurrently, adult-onset Still disease (AOSD) is also an infrequent ailment, characterized by spiking fever, arthritis, skin rash, lymphadenopathy, and serositis. Although the precise pathogenesis remains incompletely understood, some case reports suggest that ASIA may be at the root of AOSD development with the same instigator. In this context, we present three cases of patients diagnosed with AOSD, which possibly could be considered an association with ASIA, years after undergoing breast reconstruction with silicone breast implants. In one case, the patient solely received medical treatment due to her refusal to have the implant removed, resulting in multiple flares and severe complications related to glucocorticoid therapy. Conversely, in the other two cases, a combination of immunosuppressive therapy and silicone breast implant explantation led to the complete resolution of clinical symptoms. To the best of our knowledge, there are only 10 documented case reports of AOSD associated with silicone breast implants insertion. We believe this report serves as a complementary addition to prior research and offers further insights into the ongoing debate about whether explantation should be carried out early in the clinical course or not.

Publisher

Ovid Technologies (Wolters Kluwer Health)

Reference13 articles.

1. “ASIA”—autoimmune/inflammatory syndrome induced by adjuvants.;Shoenfeld;J Autoimmun,2011

2. Separating myth from reality in breast implants: an overview of 30 years of experience.;McGuire;Plast Reconstr Surg,2023

3. Preliminary criteria for classification of adult Still’s disease.;Yamaguchi;J Rheumatol,1992

4. Recognizing autoimmune/autoinflammatory syndrome induced by adjuvants (ASIA) useful knowledge for identifying patients with underlying immune dysregulation.;Kaczor;Nat Med J,2017

5. Clinical findings in symptomatic women with silicone breast implants.;Vasey;Semin Arthritis Rheum,1994

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