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間質性肺炎, 間質性腎炎を呈した潜在型1次性Sjogren症候群の1男性例: 診断に耳下腺MRIが有用であった症例

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Title: 間質性肺炎, 間質性腎炎を呈した潜在型1次性Sjogren症候群の1男性例: 診断に耳下腺MRIが有用であった症例
Other Titles: A male case of primary Sjogren's syndrome with interstitial pneumonitis and interstitial tubulo-nephritis in the absence of dry eye and dry mouth: parotid gland MRI is a useful diagnostic method for Sjogren's syndrome
Authors: 高村, 昇 / 江口, 勝美 / 右田, 清志 / 塚田, 敏昭 / 溝上, 明成 / 折口, 智樹 / 長瀧, 重信 / 泉, 雅浩 / 中村, 卓
Authors (alternative): Takamura, Noboru / Eguchi, Katsumi / Migita, Kiyoshi / Tsukada, Toshiaki / Mizokami, Akinari / Origuchi, Tomoki / Nagataki, Shigenobu / Izumi, Masahiro / Nakamura, Takashi
Issue Date: 28-Feb-1995
Publisher: 日本臨床免疫学会 / The Japan Society for Clinical Immunology
Citation: 日本臨床免疫学会会誌, 18(1), pp.123-132; 1995
Abstract: 肺門部リンパ節腫脹,間質性肺炎,間質性腎炎を合併したSjogren症候群の男性症例を経験した.症例は60歳男性で, 57歳時人間ドックで高γグロブリン血症を指摘された.主訴は全身倦怠感と発熱で当科に入院した.胸部X線とCT検査で肺門部リンパ節腫脹と間質性肺炎の所見が得られた.入院9カ月前より両眼ぶどう膜炎あり,サルコイドーシスが疑われたが, ACE, リンパ節生検と肺生検から否定された.高γグロブリン血症と抗核抗体陽性からSjogren症候群が疑われたが,眼・口腔乾燥症状はなかった. Schirmerテストとrose bengalテスト陽性から乾燥性角結膜炎があり,小唾液腺生検では単核球の著しい浸潤が認められた. 耳下腺MRI所見では, T1およびT2強調画像で多数の点状の高信号域のため不均一となっており,耳下腺組織の破壊が高度で脂肪変性をきたしていると診断した. 腎生検所見では間質・尿細管の病変が著明でいわゆるtubulo-interstitial nephritisを示した. 本症例は男性で潜在型Sjogren症候群であり,耳下腺MRI所見が本症の診断に有用であった.本検査は侵襲が少なく, Sjogren症候群の診断法の1つとして繁用されることが期待される. / Here we report a case of primary Sjogren's syndrome with hilar lymphadenopathy, interstitial pneumonitis and interstitial tubulo-nephritis. A 60-year old man was admitted to our hospital in May 1993 because of general fatigue and fever.He was noted to have hypergammaglobulinemia and had positive antibodies to nuclear antigens since 1990 in the absence of clinical manifestations. Since 9 months before admission, he presented with general fatigue, low grade fever and uveitis.On admission, chest X-ray and CT scan showed bilateral hilar lymphadenopathy and interstitial pneumonitis. The negative results for both serum angiotensin converting enzyme and histological findings of the cervical lymph node and the lung excluded the diagnosis of sarcoidosis. Serological examination exhibited marked elevation of polyclonal IgG leveland anti-nuclear antibody, but neither anti-SS-A(Ro) nor anti-SS-B(La) antibody was detected. He did not have symptoms of xerophthalmia and xerostomia. Keratoconjunctivitis sicca was diagnosed by positive Schirmer's and rose bengal tests. His labial glandbiopsy demonstrated severe mononuclear cell infiltration around the ducts. MRI findings ofthe parotid glands revealed heterogenous and dotted high signal intensity similar to those in fat tissues in the T1-and T2-weighted images. These findings depicted that bilateral parotid gland was extensively destructed and was replaced by lipid tissue. Renal biopsy showed interstitial tubulo-nephritis. On the basis of the above findings, he was diagnosed to have primary Sjogren's syndrome and uveitis. Therefore, MR image of the parotid gland is considered to be a noninvasive and useful method for diagnosis of Sjogren's syndrome.
Keywords: Sjören's syndrome / MRI findings of parotid gland / interstitial tubulo-nephritis / uveitis
URI: http://hdl.handle.net/10069/24064
ISSN: 09114300
Rights: Copyright (c) 1995 日本臨床免疫学会
Type: Journal Article
Text Version: publisher
Appears in Collections:Articles in academic journal

Citable URI : http://hdl.handle.net/10069/24064

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