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]
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,
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.
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.
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.
Kumagai Y, Ave C, Shiokawa Y. Scleroderma after cosmetic
surgery—four cases of human adjuvant disease. Arthritis Rheum 1979; 22:532-7
Van Nunen SA, Gatenby PA, Basten A. Post-mammoplasty
connective tissue disease. Arthritis Rheum
Ellenbogen R, Ellenbogen R, Rubin L. Injectable fluid
silicone therapy: Human morbidity and mortality.
JAMA 1975; 234:308-9
VargasA. Shedding of silicone particles from inflated breast implants (Letter). Plast Reconstr Surg 1979; 64:252-3
Wang PY. On the antigenicity of biomedical polymers. Advances in
biomaterials 3, Chichester: John Wiley and Sons, 1981
Pernis B. Paronetto F. Adjuvant effect of silicon (tridymite) on antibody production. Proc Soc Exp Biol Med 1962; 110:390-93
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