MEF000024040/1118

 

First page is retyped from handwritten notes, regarding 1984 article

Reviewed 1989/originally accepted for publication 1984—Case Report POST-MAMMOPLASTY HUMAN ADJUVANT DISEASE

 

In a 1973 case study a 30-year-old woman was diagnosed as having rheumatoid arthritis.  She underwent breast augmentation surgery with saline-filled Heyer-Schulte silastic envelope-type prostheses in November 1976.  Keratoconjuctivitis sicca was diagnosed in February 1982.  In December 1982 she developed skin changes overlying the left breast and later on the abdomen.  Biopsy confirmed the diagnosis of morphoea.  Investigations revealed that she had now become strongly seropositive.  The skin lesions did not change significantly over the next six months.  Previous reports of post-mammoplasty connective tissue disease have followed the use of silicone gel, whereas the more ‘biologically inert’ saline-filled implants were used in this case.  It is possible that the rheumatoid arthritis and morphoea could have occurred as a chance happening, but the development of a second connective tissue disease following implantation surgery may represent human adjuvant disease occurring in a genetically predisposed individual.

 

[End of hand written notes]

 

MEF000024040-43/1118

 

Case Report

POST-MAMMOPLASTY HUMAN ADJUVANT DISEASE

By  M.A. Byron¹, V.A. Venning² and A.G. Mowat¹

¹ Department of Rheumatology, Nuffield Orthopaedic Centre, Headington, Oxford OX3 7LD

² Department of Dermatology, Churchill Hospital, Headington, Oxford

 

Summary

Morphoea and Keratoconjuctivitis sicca developed in a woman with seronegative rheumatoid arthritis six years after augmentation mammaplasty.  Previous reports of post-mammoplasty connective tissue disease have followed the use of silicone gel, whereas, the more ‘biologically inert’ saline-filled implants were used in this case.

 

KEY WORDS:  Mammoplasty, Silicone, Adjuvant Disease, Morphoes

 

Since 1964, more than 30 cases have been described of connective tissue disease developing in patients who have previously undergone cosmetic surgery using implantations of paraffin, processed petroleum or silicone.  The majority of these reports came from Japan (1) when techniques of implantation were crude.  More recently, four cases were reported from Australia (2) following augmentation mammoplasty with silicone gel-filled elastomer envelope-type prostheses.

 

Case history

 

A 30 year-old woman was diagnosed as having rheumatoid arthritis in 1973.  Although she was strongly seronegative for rheumatoid factor, because of persistent synovitis and radiographic evidence of progressive erosions she was treated with a number of second-line agents.   Clinically she responded well to these drugs, but side-effects led to their discontinuation (Chloroquine—rash; gold—rash; levamisole—flu-like symptoms).  Since 1980 she has been maintained on nonsteroidal anti-inflammatory drugs alone.

 

In November 1976, she underwent breast augmentation surgery with saline-filled Heyer-Schulte silastic envelope-type prosthesis.  In February 1982 she first complained of grittiness of the eyes.  Keratoconjuctivitis sicca was diagnosed by a Schirmer test and Rose Bengal staining.  There was no xerostomia or parotid gland enlargement.  In December 1982 she developed changes in the skin overlying the left breast, and several weeks later noticed a similar appearance of the skin of the abdomen.  On examination, well-demarcated areas of tethered, indurated and de-pigmented skin were seen circumscribing the left breast and over the side of the abdomen.  Biopsy confirmed the diagnosis of morphoea.  There was no evidence of malignancy or infection at the sites of the prosthesis, and no clinical features of progressive systemic sclerosis.  Investigations revealed that she had now become strongly seropositive (Rose Waaler 1:4096), although no other auto-antibodies including antinuclear and parotid antibodies were found.  The skin lesions did not change significantly over the next six months.

 

COMMENT

 

Early methods of breast augmentation surgery involved the direct instillation of liquid silicone in the breasts, often with an intense local inflammatory reaction (1,2). Although the advent of elastomeric silicone envelope prostheses reduced this severe reaction, it is known that silicone particles can still ‘leak’ from such implants (4) into the surrounding tissues and to regional lymph nodes with resultant foreign body granulomatous reactions.

 

This potentially antigenic material has been implicated as an adjuvant in post-mammoplasty human adjuvant disease (2).  The development of silicone and other hydrocarbon polymers which appear to be well tolerated in animal experiments has led to the idea that these substances are biologically inert.  However, the response does not appear to be species-specific, suggesting genetic control of the immunological response (5), and the slow degradation of these polymers in vivo may account for their antigenicity.  Silicone may also provoke an autoimmune response by conversion to silica, a substance known to exert profound effects on the immune system (6), and to be associated with an increase prevalence of progressive systemic sclerosis (7).   A variety of auto-immune diseases has been described in association with cosmetic surgery, including progressive systemic sclerosis, systemic lupus erythematosus, rheumatoid arthritis, thyroiditis, Sjogren’s syndrome and morphoea (1), and is some cases there has been dramatic improvement in symptoms following bilateral mastectomy.  The rarity of this association, however, suggests that additional factors are required for the development of overt disease. By analogy with other auto-immune diseases these could include disease susceptibility genes linked to the major histocompatibility complex.  HLA typing has only been reported in a few cases (2) and no consistent haplotype was identified.  Exacerbation of pre-existing disease following silicone implantation would be difficult to define, so it is interesting to note the seroconversion observed in our patient, although without obvious exacerbation of articular disease.  We cannot exclude the possibility that the rheumatoid arthritis and morphoea could have occurred in our patient as a chance happening, but the development of a second connective tissue disease following implantation surgery may represent human adjuvant disease occurring in a genetically predisposed individual.

 

With the growing use of silicone in a variety of implants, the now rare occurrence of human adjuvant disease may become increasingly common.

 

REFERENCES

1.                  Kumagai Y, Ave C, Shiokawa Y.  Scleroderma after cosmetic surgery—four cases of human adjuvant disease.  Arthritis Rheum 1979; 22:532-7

2.                  Van Nunen SA, Gatenby PA, Basten A.  Post-mammoplasty connective tissue disease.  Arthritis Rheum 1982; 25:694-7

3.                  Ellenbogen R, Ellenbogen R, Rubin L.  Injectable fluid silicone therapy:  Human morbidity and mortality.  JAMA 1975; 234:308-9

4.                  VargasA. Shedding of silicone particles from inflated breast implants (Letter).  Plast Reconstr Surg 1979; 64:252-3

5.                  Wang PY.  On the antigenicity of biomedical polymers.  Advances  in biomaterials 3, Chichester:  John  Wiley and Sons, 1981

6.                  Pernis B. Paronetto F. Adjuvant effect of silicon (tridymite) on antibody production.  Proc Soc Exp Biol Med 1962; 110:390-93

7.                  Rodnam GP, Benedek TG, Medsger TA Jr, Cammarata RJ, The association of PSS (scleroderma) with coal-miner’s pneumoconiosis and other forms of silicosis.  Ann Intern Med 1967; 66:323-34 

 

 

Submitted 29 November 1983;  revised 12 January:  accepted 13 January 1984.

Address correspondence to Dr. M.A. Byron