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An Unusually Complicated Case of Primary Sjögren's Syndrome: Development of Transient "Lupus-type" Autoantibodies Following Silicone Implant Rejection

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www.ProHealth.com • January 8, 2004


An Unusually Complicated Case of Primary Sjögren's Syndrome: Development of Transient "Lupus-type" Autoantibodies Following Silicone Implant Rejection

To the Editor:

We describe the appearance of transient "lupus-type" autoantibodies (anti-Sm and anti-RNP) with no clinical evidence of systemic lupus erythematosus (SLE), concurrent with the rejection of a silicone wrist implant inserted 4 years previously, in a woman with a 15 year history of primary Sjögren's syndrome (pSS) who had been followed for 8 years. In addition, she had had persistent cytopenia (leukopenia and thrombocytopenia) as well as a monoclonal gammopathy. Progression of pSS to serological and then to clinical SLE has previously been documented in one patient only. In our patient these autoantibodies might have been precipitated by silicone implant rejection. The sudden occurrence of wrist pain combined with appearance of the lupus-type autoantibodies makes this probable. After surgical removal of the implant, these autoantibodies disappeared, without progression to clinical SLE. The controversial effect of silicone implant rejection on the emergence of a different pattern of autoimmunity is briefly discussed.

A 61-year-old Caucasian woman had been diagnosed with hypothyroidism at age 40. Since 1985, she had complained of increasing dryness of the mouth and eyes and recurrent submandibular gland swellings.

Dental caries, pyelitis, cystitis, and bronchitis necessitated repeated courses of antibiotics. Gammaglobulin injections (Beriglobin; Germany) were given for 3 months. She was also given steroids (prednisone 10 mg daily), chloroquine (Nivaquine), methotrexate (MTX), and sulfasalazine. MTX and steroids were discontinued in 1996. Schirmer's test was dry (positive) bilaterally (< 5 mm) and submandibular adenopathy was present. An initial mild hypogammaglobulinemia with a monoclonal peak in the mid-gammaglobulin region was detected. The serum M component was 21 g/l and serum gammaglobulin was 5 g/l (normal 6–15 g/l), confirmed by repeated estimations. Immunofixation for IgG was high positive. The gammaglobulin was of the IgG lambda type. Bone marrow showed plentiful megakaryocytes and granulocyte precursors with mild prominence of plasma cells (9%), some of which were morphologically abnormal with large, definite nucleoli. Bence-Jones proteinuria and cryoglobulins were not detected. Serum gammaglobulin concentrations were reported as normal at 8 g/l two years later.

The erythrocyte sedimentation rate remained persistently elevated (70–90 mm/h). Antinuclear antibodies were constantly demonstrated (1:160 to 1:640, speckled pattern) as were antibodies to Ro and La. Rheumatoid factor was found in 1999 (Rose-Waaler positive); rheumatoid factor Latex was 108.8 IU/ml (normal 0–39). Antibodies to dsDNA by Crithidia luciliae were negative. Antibody titers to parvovirus and to hepatitis C were negative.

Persistent leukopenia and thrombocytopenia were present. The platelet counts fluctuated to as low as 117 ´ 109/l, and the white blood cell count to as low as 2.70 ´ 109/l (normal 4–12.0). Neutropenia as low as 0.59 ´ 109/l was detected.

Two years later, a lower limb lymphocytic vasculitis was diagnosed.

In early 2002, she complained of severe pain in her right wrist, without preceding trauma. Antibodies to dsDNA by ELISA were initially detected, but on repeat testing by immunofluorescence (C. luciliae), they proved to be negative. ELISA (confirmed by immunodiffusion and Western blot) detected antibodies to Sm at a titer of 1:5120.

Antibodies to nRNP were also detected by immunoblotting. The appearance of both these antibodies was transient. Radiographs at this time showed advanced cystic degeneration of carpal bones and fragmentation of the wrist implant (Figure 1). An excision arthroplasty was performed. The pain subsided and repeated immunological testing over the next year showed total disappearance of the lupus-type autoantibodies. Laboratory investigations for measurement of antisilicone antibodies could not be undertaken at this time.



Figure 1. Radiograph of right wrist showing multiple cystic degeneration of carpal bones following fragmentation of trapezium implant performed in 1997.
HLA testing found the patient to be A24, 11, DR15, 17, B7, 50, and C2, 7.

The progression of pSS to serological and then to clinical SLE has been documented by Satoh, et al1, who described a 69-year-old Japanese woman with pSS who developed anti-Sm antibodies after a stable course of 9 years, followed by clinical SLE. Lupus associated autoantibodies (although not to Sm and RNP) following silicone breast implants have also been reported2. Other accompaniments of pSS in our patient, such as leukopenia and thrombocytopenia and indeed neutropenia (reported in 10% of patients with pSS), although previously reported, are also uncommon3,4. In our patient it was initially combined with a mild hypogammaglobulinemia, distinctly rare in this disorder, which might have been related to previous therapy with immunosuppressives such as MTX, thus possibly adding to her predisposition to recurrent infections. Garcia-Carrasco, et al5 recently investigated the frequency of hypogammaglobulinemia in a group of Spanish patients with SS, and found 8% had low IgG levels.

These investigators also found evidence of previous parvovirus infection in 35%. As the hypogammaglobulinemia in our patient was transient, it is possible that it may have been drug induced. However, the frequency of infections seemed not to decrease following normalization of the gammaglobulin levels, and because of this, the infections may have been related to the underlying SS itself (because of mucosal abnormalities seen in this condition), combined with the neutropenia and not because of immunosuppression from drugs or the hypogammaglobulinemia. Another explanation might be an occult polymorphonuclear cell dysfunction or abnormality of T cell subsets.

There was a persistent monoclonal gammopathy and a diagnosis of monoclonal gammopathy of undetermined significance was also made6. According to several authorities, this may be predictive for the future development of myeloma7.

Monoclonal gammopathies have also infrequently been documented in association with SS. However, Sugai, et al8 studied 12 Japanese patients with pSS and found a large variety of monoclonality in their group, while Broggini, et al9 in 358 patients with SS found this to be present in 6% of patients.

Our patient, in addition, also had hypothyroidism. She thus represents another example of the "mosaic of autoimmunity" as described by Shoenfeld and Isenberg10.

The question arises as to the relationship of the silicone implant leakage and its probable rejection and the transient development of these autoantibodies. This case is not dissimilar to our report11 of a 45-year-old woman who, upon inhalation of a polyclonal lymphocyte-activating factor, developed 6 different autoimmune diseases associated with a panoply of autoantibodies. The combination of an environmental factor and a genetic predisposition is therefore well known as leading to an overt autoimmune disease.

In our case the silicone may have acted as an "adjuvant". Indeed, injections of silicone to MRL/lpr strains of mice have been followed by increased titers of SLE autoantibodies as well as cytokine changes12. Illnesses resembling rheumatoid arthritis, SLE, and SS, termed "siliconosis," following silicone breast augmentation have been reported13, as well as a fibromyalgia/chronic fatigue syndrome.

Infections such as myocobacterial, gram-negative14, and specifically Epstein-Barr virus15 are also known to be associated with the emergence of SLE related autoantibodies and even overt SLE.

The transient appearance of these antibodies simultaneous to the silicone transplant rejection, and their disappearance with its speedy removal, makes a strong case for not considering such transplants in patients with preexisting autoimmune disease or diathesis.

RONALD A. ASHERSON, MD, FRCP, Rheumatic Diseases Unit, University of Cape Town Health Sciences Center, Groote Schuur Hospital, Cape Town and The Rosebank Clinic, Johannesburg, South Africa; YEHUDA SHOENFELD, MD, FRCP, Center for Autoimmune Diseases, Department of Medicine B, Sheba Medical Center, Tel-Hashomer, Israel; PETER JACOBS, BM, BCh, MD, PhD, Department of Haematology and Bone Marrow Transplantation Unit, Constantiaberg Medi Clinic, Cape Town, South Africa; CHRIS BOSMAN, MB, FCP(SA), The Kenridge Hospital, Johannesburg, South Africa.



REFERENCES

1. Satoh M, Yamagata H, Watanabe F, et al. Development of anti-Sm and anti-DNA antibodies followed by clinical manifestation of systemic lupus erythematosus in an elderly woman with long-standing Sjogren's syndrome. Lupus 1995;4:63–5.

2. Zandman-Goddard G, Blank M, Ehrenfeld M, Gilburd B, Peter J, Shoenfeld Y. A comparison of autoantibody production in asymptomatic and symptomatic women with silicone breast implants. J Rheumatol 1999;26:73-7.

3. Schattner A, Friedman J, Klepfish A. Immune cytopenias as the presenting manifestation of primary Sjogren's syndrome. QJM 2000;93:825-9.

4. Richert-Boe KE. Hematologic complications of rheumatic disease. Hematol Oncol Clin North Am 1987;1:301-20.

5. Garcia-Carrasco M, de la Red G, Ramos-Casals M, et al. Low positivity of anti-Ro/SS-A and anti-La/SS-B in patients with primary Sjogren's syndrome and hypogammaglobulinaemia [abstract]. Autoimmun Rev 2002;1:51.

6. Kyle RA, Lust JA. Monoclonal gammopathies of undetermined significance. Semin Hematol 1989;26:176-200.

7. Terpos E, Angelopoulou MK, Variami E, Meletis JC, Vaiopoulos G. Sjogren's syndrome associated with multiple myeloma. Ann Hematol 2000;79:449-51.

8. Sugai S, Shimizu S, Tachibana J, et al. Monoclonal gammopathies in patients with Sjogren's syndrome. Jpn J Med 1988;27:2-9.

9. Broggini M, Cavallo A, Baratelli E, et al. Monoclonal gammopathy of uncertain significance in rheumatic disease. Recenti Prog Med 1990;81:306-9.

10. Shoenfeld Y, Isenberg D. The mosaic of autoimmunity; Research monographs in immunology. Vol 12. Amsterdam: Elsevier Science Publishers; 1989.

11. Rahamim-Cohen D, Shoenfeld Y. The mosaic of autoimmunity. A classical case of inhalation of a polyclonal activating factor in a genetically and hormonally susceptible patient leading to multiple autoimmune diseases. Isr Med Assoc J 2001;3:381-2.

12. Schaefer CJ, Wooley PH. The influence of silicone implantation on murine lupus in MRL lpr/lpr mice. J Rheumatol 1999;26:2215-21.

13. Borenstein D. Siliconosis: a spectrum of illness. Semin Arthritis Rheum 1994;24 Suppl 1:1-7.

14. Isenberg DA, Maddison P, Swana G, et al. Profile of autoantibodies in the sera of patients with tuberculosis, Klebsiella and other gram-negative infections. Clin Exp Immunol 1987;67:516-23.

15. James JA, Neas BR, Moser KL, et al. SLE in adults is associated with previous EBV exposure. Arthritis Rheum 2002;44:1122-6.

Source: Journal of Rheumatology.

© 2004. The Journal of Rheumatology Publishing Company Limited.
All rights reserved.



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