Katsumi SUGIURA, M.D., Mamoru TANI, M.D., Munenobu MUYA, M.D., Masaaki ISONOKAMI, M.D., Noboru TANABE, M. D., Tomohito HONDA, M. D. and Takehito KOZUK
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1 Katsumi SUGIURA, M.D., Mamoru TANI, M.D., Munenobu MUYA, M.D., Masaaki ISONOKAMI, M.D., Noboru TANABE, M. D., Tomohito HONDA, M. D. and Takehito KOZUKA, M.D.
2 Fig. 1 Infiltation of More than 50 Mononuclear Cells around Intralobular Fig. 2 Hyaline Necrosis of Subcutaneous Fat (H. E. stain ~ 40) Ducts (H. E. stain ~ 200) Fig. 3 Lymphoid Follicles with Germinal Center in Subcutaneous Fat (H. E. stain ~ 40) Fig. 4 Lymphocytic Vasculitis and Thrombosis in Deep Dermis (H.E. stain ~ 40)
3 Table 1 Diagnostic Criteria for Sjogren's Syndrome') Definite Presence of sicca complex symptoms of unknown case, together with one or more of the following three features; 1. evidence of idiopathic KCS: (++) or over response in the Rose Bengal staining, and the wetting of less than 10mm in the Shirmer test, or a positive fluorescein staining. 2. characteristic pathologic findings within lacrimal and/or salivary glands: infiltration of more than 50 mononuclear cells aroud intralobular ducts, found in at least one spot within a single lobule. 3. definite pathologic findings on sialography: findings of diffuse punctate shadows with diameters of more than 1 mm Probable Presence of sicca complex symptoms of unknown cace, together with one or more of the following three features: 1. suspicion of idiopathic KCS: (+) response in the Rose Bengal staining, and the wetting of less Case Case than 10mm in the Shirmer test, or a postive fluorescein staining. 2. evidence of reduced salivary secretion: less than 10ml of saliva, after 10 minute gum test. 3. swollen salivary glands of unknown cause, progressing recurrently of chronically
4 Table 2 Primary Criteria for the Classification of SjOgren's Syndrome 2) 1. Ocular symptoms Definition. A positive response to at least of the following 3 questions: (a) Have you had daily, persistent, troublesome dry eyes for more than 3 months? (b) Do you have a recurrent sensation of sand or gravel in the eyes? (c) Do you use tear substitutes more than 3 times a day? 2. Oral symptoms Definition. A positive response to at least 1 of the following 3 questions: (a) Have you had a daily feeling of dry mouth for more than 3 months? (b) Have you had recurrent or persistently swollen salivary glands as an adult? (c) Do you frequently drink liquids to aid in swallowing dry foods? 3. Ocular signs Definition. Objective evidence of ocular involvement, determined on the bases of apositive result on at least 1 of the following 2 tests: (a) Schirmer-I test ( 5mm in 5 minutes) (b) Rose bengal score ( 4, according to the van Bijsterveld scoring system) 4. Histopathologic features Definition. Focus score 1 on minor salivary gland biopsy (focus defined as an agglomeration of at least 50 mononuclear cells; focus score defined as the number of foci 4 mm2 of gladular tissue). 5. Salivary gland involvement Definition. Objective evidence of salivary gland involvement, determined on the basis of a positive result on at least 1 of the following 3 tests: (a) Salivary scintigraphy (b) Palotid sialography (c) Unstimulated salivary flow ( 1.5ml in 15 minutes) 6. Autoantibodies Difinition. Presence of at least 1 of the following serum autoantibodies: (a) Antibodies to Ro/SS-A or La/SS-B antigens (b) Antinuclear antibodies (c) Rheumatoid factor Exclusion criteria: preexisting lymphoma, acquired immuno-deficiency syndrome, sarcoidosis, or graft-versus-host disease
5 Table 3 Score of Parotid Sialography 3) Table 4 Histologic Findings of Lupus Erythematosus Profundus 5) Table 5 Histologic Findings of Lupus Erthematosus Profundus 6)
6 Table 6 Classification of Lupus Erythematosus Profundus Complicated by SLE or DLE Reported in Japan SjOgren's syndrome in Japan and United States, Rheum Dis Clin North Am, 18: , Vitali C, Bombardieri S, Moutsopoulos HM et al: Preliminary criteria for the classification of 347, Rubin H, Holt JF: Secretary sialography in diseases of the major salivary glands, Am J Roentgenol, 77: , Irgang S: Lupus erythematosus profundus, Arch Dermatol Syph, 42: , Winkelmann RK, Peters MS: Lupus panniculi- tis, Dermatol Update: , Peters MS, Daniel Su WP Lupus erythematosus panniculitis, Med Clin North Am, 73: , 1989 SjOgren's syndrome, Arthritis Rheum, 36: Saito I, Nishimura S, Tsubota K, Miyasaka N: Comparison of diagnosis and treatment of
7 A Case of Lupus Erythematosus Profundus with Sjogren's Syndrome Katsumi Sugiura, Mamoru Tani, Munenobu Muya, Masaaki Isonokami, Noboru Tanabe, Tomohito Honda and Takehito Kozuka Department of Dermatology, Osaka National Hospital , Hoenzaka, Cyuou-ku, Osaka 540, Japan Key words: lupus erythematosus Profundus \Sjogren's syndrome A 37-year-old woman had ocular and oral dryness with a tender subcutaneous tumor on the left hip for 2 months. Skin biopsy showed panniculitis with lymphoid follicles and with hyaline necrosis. This finding suggested to be lupus erythematosus profundus. She was diagnosed as Sjogren's syndrome by lip biopsy and sialography. Complication of lupus erythematosus profundus and collagen diseases was briefly reviewed.
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