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Autoimmune Syndrome Induced by Adjuvants (ASIA) after Silicone Breast Augmentation Surgery

Nunes e Silva, Daniel, MD*; Gründler, Cíntia, MD; Spengler, Maria das Graças de Melo Teixeira, MD; Horimoto, Alex Magno Coelho, MD§; Machado, Mariana Albuquerque, MD; Frazão, Isadora Carvalho, MD; Takita, Luiz Carlos, MD**

Plastic and Reconstructive Surgery – Global Open: September 2017 - Volume 5 - Issue 9 - p e1487
doi: 10.1097/GOX.0000000000001487
Case Report
Brazil

Summary: Generally, the main complications of silicone implantation are local symptoms. However, some patients develop late-onset systemic symptoms often associated with a rare form of hyperactive immune response, as part of a syndrome known as autoimmune syndrome induced by adjuvants (ASIA). Reported cases of ASIA have shown resolution with explantation, but not with immunomodulatory therapy. In this report, we described a case of a previously healthy 23-year-old woman, who has undergone silicone breast implant augmentation, for aesthetic reasons, and developed localized cutaneous impairment 3 years postsurgery. She received a diagnosis of ASIA with a new presentation: Lupus-like manifestation through localized cutaneous impairment. This patient’s symptoms were managed without the need for surgical intervention, which has not been previously reported, because the patient did not want an explantation for aesthetic reasons. The patient was started on hydroxychloroquine, 400 mg per day, and remains asymptomatic after 2 years of treatment. The exact predisposition to ASIA is still unknown. Without implant explantation and with immunomodulatory treatment, this patient’s condition substantially improved. Based on our current understanding of this disease, it might not be prudent to indicate breast augmentation with silicone implants in patients with documented autoimmune reaction to an adjuvant, an established autoimmune condition, or genetic predisposition. However, if a patient does develop silicone-induced ASIA, explantation is no longer the only successfully reported option, as these symptoms can be managed with immune suppression.

From the *Universidade Federal de Mato Grosso do Sul (UFMS) and UNIDERP; Campo Grande (MS), Brazil; Brazilian Society of Dermatology, Campo Grande (MS), Brazil; Brazilian Society of Dermatology, UNIDERP, Campo Grande (MS), Brazil; §Universidade Federal de Mato Grosso do Sul (UFMS), Campo Grande (MS), Brazil; Plastic Surgery Service of UFMS, Campo Grande (MS), Brazil; Plastic Surgery Service of UFMS, Campo Grande (MS), Brazil; and **Universidade Federal de Mato Grosso do Sul, Campo Grande (MS), Brazil.

Received for publication May 5, 2017; accepted July 19, 2017.

Disclosure: The authors have no financial interest to declare in relation to the content of this article. The Article Processing Charge was paid for by the authors.

Daniel Nunes e Silva, MD, PhD, Federal University of Mato Grosso do Sul—UFMS, Unnumbered, Av. Costa e Silva, Cidade universitária, Mato Grosso do Sul 79070–900, Brazil, E-mail: danielnunesesilva@gmail.com

This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal.

From the 1990s, some patients who had silicone breast augmentation surgery presented with a collection of symptoms that did not fit the criteria for any defined connective tissue disorder, suggesting that a nondefined syndrome may appear after the exposure to silicone in plastic surgery.1

Autoimmune syndrome induced by adjuvants (ASIA) is an autoimmune syndrome that characterizes many of these cases.1 Shoenfeld and Agmon-Levin2 defined a set of major and minor criteria to diagnose ASIA (Table 1). Patients must show, at least, 2 major criteria or 1 major and 2 minor criteria.

Table 1

Table 1

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CASE REPORT

A 23-year-old Caucasian, Brazilian woman, with breast augmentation performed 3 years prior, presented with a progressive breast erythema. She underwent a bilateral silicone breast implantation (SBI) with retroglandular polyurethane silicone gel prostheses (275 ml, Silimed) inserted through inframammary incisions. The patient’s perioperative course was uneventful (Figs. 1, 2). She has no medical history or known allergies; also, she did not take any medications. During the first 3 years following the procedure, she had not reported any abnormality. The patient denied nude sunbathing.

Fig. 1

Fig. 1

Fig. 2

Fig. 2

During her scheduled 3-year postsurgery follow-up, she mentioned recent erythematous, progressively growing and nonpruritic lesion that appeared on her breasts. Her physical examination was normal, except for the bilateral breast papular erythematous lesion presentation. This dermatological alteration was specifically on her breast area. There was no mammary contracture or any other abnormalities (Fig. 3).

Fig. 3

Fig. 3

The diagnostic hypothesis of allergic contact dermatitis was strongly suspected, and an allergy patch test was performed. The result was negative.

The following laboratory examinations were ordered: liver function tests, urea and creatinine, blood cell count, rheumatological tests [antinuclear antibody, anti-Smith, anti-Sjögren’s syndrome type B, anti-Sjögren's-syndrome-related antigen A, anti-nuclear ribonucleoprotein, lupus anti-coagulant, anti-double stranded DNA, anti-immunoglobulin G (IgG) and IgM cardiolipin, and erythrocyte sedimentation rate], glucose, thyroid hormones, active hepatitis B and C, syphilis, and HIV. The patient reported a positive antinuclear antibody, with a titer of 1:640, presenting a nuclear speckled pattern. The remaining tests were negative.

An ultrasonography and magnetic resonance imaging were performed and did not show any abnormalities. The mammary implants were intact.

A skin biopsy of an erythematous area on her left breast was performed and indicated stretches of thinning epidermis, focal hyperkeratosis, basal vacuolar degeneration, and thickening of the epithelial basement membrane. The papillary and superficial dermis showed a perivascular lymphomononuclear infiltrate. Irritative or allergic contact dermatitis, neoplasia, or presence of fungi were excluded. Direct immunofluorescence showed dense granular continuous deposits of IgG (+++) along the dermoepidermal junction (lupus band test) (fluorescein isothiocyanate anti-IgG).

From her clinical findings and laboratorial results, and since the case did not fit all diagnostic criteria for systemic erythematous lupus, the patient was diagnosed with silicone-induced ASIA—with a lupus-like manifestation through localized cutaneous impairment.

Therefore, the patient was started on hydroxychloroquine, 400 mg per day. There was a progressive improvement of lesions after 3 months, and no lesions were present after 6 months. She still receives hydroxychloroquine and remains asymptomatic after 2 years (Fig. 4).

Fig. 4

Fig. 4

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DISCUSSION

Although rare, silicone-induced ASIA cases are becoming increasingly frequent.3 The localized cutaneous involvement and the absence of other clinical manifestations reinforce the novelty of the case. In addition, the patient’s rapid and complete response to clinical treatment, in conjunction with her clinical presentation and the integrity of the implants, avoided an explantation, as the first option, which was not the patient’s wish, for aesthetic reasons.

The patient was diagnosed with silicone-induced ASIA for presenting with 2 of the major diagnostic criteria: the exposure to external stimuli (SBI) and the typical biopsy of involved organ, and for presenting with 2 minor diagnostic criteria: presentation of autoantibodies and a clinical manifestation (cutaneous impairment).

Cutaneous manifestations located in the breasts, precisely in the detached areas for implant placement, have not yet been found in the literature. Based on current knowledge, it might not be prudent to indicate breast augmentation with silicone implants in patients with documented autoimmune reaction to an adjuvant, established autoimmune conditions, and a genetic predisposition.3 The proposed algorithm to ASIA management is displayed in Figure 5.

Fig. 5

Fig. 5

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CONCLUSIONS

The exact predisposition to ASIA is still unknown. Our reported case highlights a rare local manifestation of ASIA, successfully treated with immune suppressive therapy, thereby avoiding implant explantation, which was required in all cases reported previously in literature. Moreover, there was no recurrence of symptoms in 2 years of follow-up. Therefore, it suggests that silicone is a weak antigenic material and still can be preserved in rare ASIA cases with appropriate intervention.

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REFERENCES

1. Maijers MC, de Blok CJ, Niessen FB, et al. Women with silicone breast implants and unexplained systemic symptoms: a descriptive cohort study. Neth J Med. 2013;71:534–540.
2. Shoenfeld Y, Agmon-Levin N. ‘ASIA’—autoimmune/inflammatory syndrome induced by adjuvants. J Autoimmun. 2011;36:4–8.
3. Goren I, Segal G, Shoenfeld Y. Autoimmune/inflammatory syndrome induced by adjuvant (ASIA) evolution after silicone implants. Who is at risk? Clin Rheumatol. 2015;34:1661–1666.
Copyright © 2017 The Authors. Published by Wolters Kluwer Health, Inc. on behalf of the American Society of Plastic Surgeons. All rights reserved.