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Labial salivary gland lymphocytic infiltration in sjgren's syndrome.

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scored as positive if the neutrophils showed positive fluorescence at a serum dilution of at least 1:20. Sera were also
analyzed for the presence of autoantibodies to MPO. An
enzyme-linked immunosorbent assay using MPO as the
antigen and an MPO-specific monoclonal antibody was performed as described elsewhere (3,4).
Sera from 13 of the 75 RA patients were positive for
perinuclear ANCA (pANCA) of the IgG class. Six of these
13 patients had vasculitis (Table I). None of the patients had
Felty’s syndrome or neutropenia. Four patients had renal
involvement; a renal biopsy was performed on 1 of them.
Five of the 6 patients had MPO autoantibodies. One of these
5 patients developed pulmonary hemorrhage and glomerulonephritis (Goodpasture’s syndrome), without antibodies to
renal glomerular basement membrane, during treatment with
D-penicillamine. The patient was treated with corticosteroids, cytostatic drugs, and plasmapheresis, with good results. Over the course of followup, the ANCA and autoantibodies to MPO disappeared.
We have thus observed the possible association of
pANCA with RA. We agree with Cohen Tervaert et al that a
perinuclear staining pattern is nonspecific for antimyeloperoxidase antibodies. Other antibodies, such as granulocytespecific antinuclear antibodies, can also produce this pattern, and these antibodies have been found in serum and
synovial fluid of patients with RA ( 5 ) . However, autoantibodies to MPO can be associated with vasculitis in the
presence of RA. To our knowledge, there are no previous
reports of the finding of pANCA, and particularly, of MPO
antibodies, in a patient with pseuddoodpasture’s syndrome induced by treatment with D-penicillamine.
Slim Lassoued, MD
Centre Hospitalier General
Cahors, France
Laurent Sixou, MD
FranGoise Oksman, MD
Marc Pages, MD
Antoine Fournie, MD
Centre Hospitalier Universitaire
Toulouse. France
1. Cohen Tervaert JW, Goldschmeding R, Elema JD, Limburg PC,
Table 1. Clinical and laboratory findings in patients with rheumatoid arthritis and ANCA*
* Sera from 75 patients were tested for antineutrophil cytoplasmic
antibodies (ANCA) by indirect immunofluorescence; 13 showed
perinuclear positivity and were further tested for myeloperoxidase
(MPO) autoantibodies (see text for details). Patient 12 developed
Goodpasture’s syndrome (GPS) during treatment with D-penicillamine. R F = rheumatoid factor; ANA = antinuclear antibodies;
CIC = circulating immune complexes; hemat. = hematuria; FSG =
focal and segmental glomerulonephritis.
van der Giessen M, Huitema MG, Koolen MI, Hen6 RJ, The TH,
van der Hem GK, von dem Borne AEGKr, Kallenberg CGM:
Association of autoantibodies to myeloperoxidase with different
forms of vasculitis. Arthritis Rheum 33: 1 2 6 1 2 7 2 , 1990
Van der Woude FJ, Rasmussen N, Lobatto S , Wiik A, Permin H,
van Es LA, van der Giessen M, van der Hem GK, The TH:
Autoantibodies to neutrophils and monocytes: a new tool for
diagnosis and marker of disease activity in Wegener’s granulomatosis. Lancet I:425429, 1985
Falk RJ, Jennette JC: Anti-neutrophil cytoplasmic autoantibodies with specificity for myeloperoxidase in patients with systemic
vasculitis and idiopathic necrotizing and crescentic glomerulonephritis. N Engl J Med 318:1651-1657, 1988
Rasmussen N , Sjolin C, Isaksson B, Bygren P, Wieslander J: An
ELISA for the detection of anti-neutrophil cytoplasm antibodies
(ANCA). J Imrnunol Methods 127:139-145, 1990
Wiik A, Jensen E , Friis J: Granulocyte-specific antinuclear
factors in synovial fluids and sera from patients with rheumatoid
arthritis. Ann Rheum Dis 33515-522, 1974
Labial salivary gland lymphocytic infiltration in
Sjogren’s syndrome
To the Editor:
We read with interest the article by Shah et al, on
labial salivary gland (LSG) histopathology and connective
tissue disease (CTD) (1). The authors conclude that LSG
biopsy has become a mainstay in the diagnosis of Sjogren’s
syndrome. However, they found that lymphocytic infiltrates
seen on LSG biopsy were associated more strongly with the
presence of antinuclear antibodies than with Schirmer test
results or parotid gland enlargement, and the association of
LSG histopathologic findings with sicca symptoms was even
weaker than their association with keratoconjunctivitis sicca
by Schirmer test or with glandular enlargement. Surprisingly, the relationship between salivary gland biopsy findings
and lacrimal gland function was investigated, whereas functional studies of the salivary glands were not reported. The
authors conclude their report by suggesting the initiation of
multicenter studies on the prevalence and clinical implications of lymphocytic infiltration of the salivary glands.
For decades, investigators have noted a discrepancy
between the histopathologic features and the function of the
salivary glands (see refs. 2 and 3; for review, see ref. 4).
However, this observation has thus far received little attention. Over the last decade, we have been conducting prospective (immuno-)histopathologic and functional studies of
the salivary glands in various diseases, including CTD. Our
results have clearly confirmed the previous observations and
have further demonstrated the nonspecificity (5-8) and the
dynamic nature (9) of LSG lymphocytic infiltrates, including
the lack of correlation between lymphocytic infiltration of
the LSG and salivary gland function (5,6,9,10). It has been
shown as a secondary finding in extended studies that
lymphocytic infiltration of the salivary glands seems to be a
common phenomenon, occurring in highly diverse conditions including CTD, not implying present or future functional impairment (5-9).
The time has come to include this acquired knowledge about the lack of specificity of LSG lymphocytic
infiltration, and its low level of association with clinical
features of Sjogren’s syndrome, in any discussion of diagnostic criteria for the disease. As has already been suggested
(1 l ) , objectively verified functional impairment of exocrine
glands, in a setting of autoimmunity (defined broadly), seems
to epitomize what most of us perceive as Sjogren’s syndrome. The circle is thereby closed, as this was the description initially proposed by Henrik Sjogren in his thesis
published in 1933 (12).
At the present stage of knowledge, LSG biopsy
seems to be of little use except for scientific purposes. When
unequivocal symptoms and signs of oral and ocular sicca are
present, the presence of lymphocytic infiltrates seen on LSG
biopsy adds little to the diagnosis of Sjogren’s syndrome.
When the diagnosis is in doubt, a positive biopsy result does
not differentiate between Sjogren’s syndrome and other
connective tissue diseases.
G. Lindahl, MD, PhD
E. Hedfors, MD, PhD
Huddinge University Hospital
Karolinska Institute
Stockholm. Sweden
1. Shah F, Rapini RP, Arnett FC, Warner NB, Smith CA: Association of labial salivary gland histopathology with clinical and
serologic features of connective tissue diseases. Arthritis
Rheum 33:1682-1687, 1990
2. Albrich K: Die Keratitis filiformis und die Sekretion der Tranendrusen. Arch Ophthalmol 121:402-410, 1928
3. Fox Rl, Howell FV, Bone RC, Michelson P: Primary Sjogren’s
syndrome: clinical and immunopathologic features. Semin Arthritis Rheum 14:77-105, 1984
4. Lindahl G: Studies on the immunohistopathology of the lip
salivary glands in connective tissue diseases (thesis). Karolinska Institute, Stockholm, Sweden, 1988
5. Lindahl G, Lefvert A-K, Hedfors E: Periductal lymphocytic
infiltrates in salivary glands in myasthenia gravis patients lacking Sjogren’s syndrome. Clin Exp Immunol 66:95-102, 1986
6. Lindahl G, Hedfors E: Lymphocytic infiltrates and epithelial
HLA-DR expression in lip salivary glands in connective tissue
disease patients lacking sicca: a prospective study. Br J Rheumatol 28:293-298, 1989
7. Hedfors E, Lindahl G: Studies on the specificity of the immunohistopathological changes of the salivary glands in Sjogren’s
syndrome. Clin Exp Rheumatol7: 163-165, 1989
8. Hedfors E: The clinical use of immunohistopathology (editorial). Br J Rheumatol 28:278-280, 1989
9. Lindahl G, Lonnquist B, Hedfors E: Lymphocytic infiltration
and HLA-DR expression of salivary glands in bone marrow
transplant recipients: a prospective study. Clin Exp Immunol
721267-273, 1988
10. Hedfors E, Lindahl G: The occurrence of MHC class I and I1
antigens in relation to lymphocytic infiltrates and interferon-g
positive cells in lip salivary glands. J Rheumatol 17:743-750,
11. Fox Rl, Robinson CA, Curd JG, Kozin F, Howell FV: Sjogren’s
syndrome: proposed criteria for classification. Arthritis Rheum
12. Sjogren H: Zur kenntnis der Keratoconjunctivitis sicca. Acta
Ophthalmol [Copenhl [Suppl] 2: 1-151, 1933
Monozygotic twins concordant for Behset’s syndrome
To the Editor:
Factors that suggest a genetic predisposition to BehGet’s syndrome (BS) are the association with HLA-B5, the
occasional reports of familial involvement, and an ethnic
aggregation, with greater prevalence among the people of the
Mediterranean basin and Japan (1,2).
Concordance in monozygotic twins would be another
clue to a genetic predisposition for BS, although we are not
aware of any reports of either the concordant or discordant
occurrence of BS in monozygotic twins. We have recently
treated 2 brothers with BS who are monozygotic twins and
have been raised together, living in the same environment.
The brothers were born in 1965, and both of them developed
BS at age 23. The other members of their family are reported
to be healthy.
Patient 1 began to experience recurrent oral ulcerations in January 1988, which was followed by the development of scrotal ulcers, ostiofolliculitis, and erythema nodosum-like lesions on his legs. In June 1988, he came for
treatment at our center because of iridocyclitis of both his
eyes. Physical examination at that time showed oral and
scrotal ulcers, with scrotal scarring, ostiofolliculitis, and
erythema nodosum-like lesions. Ophthalmologic examination revealed vitreal cells and retinal vasculitis in both fundi.
Visual acuity in both eyes was limited to “finger counting.”
The pathergy reaction (the nonspecific hypersensitivity of
the skin to a needle prick [31) was positive. Azathioprine was
Patient 2 began to experience recurrent oral and
scrotal ulcers, with ostiofolliculitis, in May 1988. One year
later, eye involvement began, and he sought treatment at our
center. Ophthalmologic examination revealed vitreal cells in
both eyes, but retinal involvement only in the right eye.
Visual acuity was 3/10 in the right eye, and 10/10 (perfect
vision) in the left eye. The pathergy reaction was positive.
Azathioprine therapy was begun. Erythema nodosum and
retinal involvement of this patient’s left eye have not yet
The obvious similarities of their physical features
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syndrome, labial, sjgren, gland, infiltrating, salivary, lymphocytes
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