有痛性筋症状を呈し，シェーグレン症候群を合併したサルコイドーシスの１例

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1 1 有痛性筋症状を呈し, シェーグレン症候群を合併したサルコイドーシスの 1 例要旨 54 1 X 2 1 キーワード : A Case of Sarcoidosis with Symptomatic Muscle Involvements and Sjögren s Syndrome Shoko Matsui, Hidehiko Aikawa, Naohiro Yamashita, Nobuki Arai, Hirofumi Taki, Eiji Sugiyama, Muneharu Maruyama, and Masashi Kobayashi ABSTRACT A 54-year old woman was admitted to a local hospital with severe muscle pain and solid masses of both legs. She was transfered to our hospital for further examination, because sarcoidosis was suspected by the histological examination of muscle biopsy. A chest X-ray film revealed bilateral hilar lymphadenopathy and diffuse reticulonodular opacities in both lungs. Histological examination of transbronchial lung biopsy specimens showed non-caseous epithelioid cell granulomas, and on these bases a diagnosis of sarcoidosis was made. Her clinical conditions improved, and solid masses subsided after the administration of prednisolone. She had the symptoms of eye and mouth dryness for 2 years, and the diagnosis of Sjögren s syndrome was made by laboratory findings and sialography. This was a rare case of sarcoidosis with symptomatic muscle involvements and Sjögren s syndrome. keywords ; Sarcoidosis, Muscle nodules, Steroid therapy, Sjögren s syndrome 富山医科薬科大学医学部第一内科著者連絡先 : 松井祥子富山医科薬科大学第一内科富山市杉谷 2630 TEL FAX First Department of Internal Medicine, Toyama Medical and Pharmaceutical University, Faculty of Medicine 65

2 日サ会誌 2000,20(1) はじめに 1) 2) SS 3) SS 症例患者 54 主訴既往歴家族歴現病歴入院時現症 155cm 57kg /88mmHg 70/ II VI 3cm 10 5cm 入院時検査所見 Table 1 - GTP ALP ACE 30U/ml SS-A C1Q-IC 3.2 GEQ/ dl X BHL II Figure 1.A CT Figure 1.B MRI rt. vastus medialis muscles T1 iso high T2 highintensity cm 9cm Figure 2.A Ga Figure 2.B Table.1 Laboratory findings on admission Hematology ESR 11 mm/h WBC 4250/mm 3 CRP 0.1 mm/h Neutro 34.0% Serology Lymph 45.0% RF 44 IU/ml Mono 11.0% ANA 2.0 U/ml Eo 7.0% SS-A x16 RBC /mm 3 SS-B (-) Hb 12.2 g/dl C1q-IC 3.2 GEQ/dl Plt /mm 3 C3d-IC 4.4 GEQ/dl Blood chemistry Blood gas analysis (Room air) TP 7.2 g/dl ph Alb 63.0% PaCO Torr 1 Grob 2.4% PaO Torr 2 Grob 8.2% Hco mmol/l Grob 8.8% BE 3.6 mmol/l Grob 17.6% Pulmonary function test GOT 26 IU/l FVC 2.55 l GPT 15 IU/l %VC 100.7% LDH 207 IU/l FEV l ALP 341 IU/l FEV 1 % 81.1% -GTP 56 IU/l %DLco 78% Amy 110 IU/l BALF CPK 140 IU/l TCC /ml ACE 30 IU/l recovery 51% Lyso 14.3 g/ml M 71.7% PPD (-) Lymph 28.3% CD4/

1 CD4/CD8 Figure 3.A Figure 3.B polyphasic discharge (Figure 1.B) (Figure 1.

B) Figure 1 Chest X-ray film (A) and Chest CT scan (B) taken on December 16, 1996,

3 1 CD4/CD8 Figure 3.A Figure 3.B polyphasic discharge (Figure 1.B) (Figure 1.A) 2 SS-A Schirmer SS SS (Figure 2.A) (Figure 2.B) Figure 1 Chest X-ray film (A) and Chest CT scan (B) taken on December 16, 1996, showing hilar and mediastinal lymphadenopathy in both lungs and reticulonodular shadows in the whole lung fields. Figure 2 Right femoral MRI showing a giant nodule in the femoral muscle (A; indicated by arrows). Gallium-67 scintigram showing marked accumulations in the mediastinum of the lungs and both legs (B). (Figure 3.A) (Figure 3.B) Figure 3 Photomicrograph of a transbronchial lung biopsy specimen from the rt. S5 (A), showing non-caseating epithelioid cell granuloma and Langhans giant cells (HE stain x15). Photomicrograph of a muscle biopsy specimen in the right lower leg (B), showing giant noncaseating granuloma with degenerative changes in the muscle fibers (HE stain x15). 67

日サ会誌 2000,20(1) 入院後経過 60mg/ 1 ACE X CT BHL MRI Figure 4.A Ga Figure 4.B 4 2 7.5mg/ (Figure 4.A) (Figure 4.

Gallium-67 scintigram showing no abnormal accumulation in the lungs and both legs (B).

4 日サ会誌 2000,20(1) 入院後経過 60mg/ 1 ACE X CT BHL MRI Figure 4.A Ga Figure 4.B mg/ (Figure 4.A) (Figure 4.B) Figure 4 Right femoral MRI obtained after corticosteroid therapy, showing disappearance of nodules in the muscle (A), and Gallium-67 scintigram showing no abnormal accumulation in the lungs and both legs (B). 考察 5) myopathy 1) Zisman % 75% 0.5% 6) myopathy ) 7) myopathy 8) myopathy 30mg/ 60mg/ 2 7.5mg/ SS 9-12) sicca 13 SS SS CD4 TIFN- IL-2mRNA 3 CD4 TIFN- IL-2 Th1 68

5 1 Immune complex SS 引用文献 1) 山本正彦 : 筋病変. 日本サルコイドーシス学会編. 最近のサルコイドーシス. 現代医療社, 東京,1993; ) 栢沼勝彦, 宇尾野公義 : 仮性筋肥大を呈した腫瘤型筋サルコイドーシスの1 例. 臨床神経学 1987; 27: ) Anaya J-M and Talal N : Sjögren s syndrome and connective tissue diseases associated with other immunologic disorders.in Koopman WJ ed, Arthritis and Allied Conditions. 13th ed, Williams & Wilkins, Maryland, 1997; ) 石田啓一郎, 小林理, 江部達夫 : 著明な筋肉内腫瘤を形成したサルコイドーシスの1 例. 日胸 1990; 49: ) Meyers GB, Gottlieb AM, Mattman PE, et al : Joint and skeleta muscle manifestations in sarcoidosis. Am J Med 1952; 12: ) Zisman DA, Biermann JS, Martinez FJ et al : Sarcoidosis presenting as a tumorlike muscular lesion. Medicine 1999; 78: ) 立花暉夫 : サルコイドーシスの全国臨床統計. 日本臨床 1994; 52: ) 南英五郎, 早原敏之, 高橋茂他 : 慢性のミオパチーを呈したサルコイドーシス. 神経内科 1986; 24: ) 香宗我部滋, 藤田穣, 今井龍雄他 : サルコイドーシスを合併した原発性シェーグレン症候群の1 症例. リウマチ 1992; 32: ) 秋山雄次, 鈴木輝彦, 田中政彦他 : Sjögren s 症候群を合併した両側腎結石を有するsarcoidosisの1 例. アレルギー 1992; 41: ) 桑田博史, 千田金吾, 須田隆文他 : シェーグレン症候群を合併したサルコイドーシスの 2 例. サルコイドーシス / 肉芽腫性疾患 ( 日サ会誌 ) 1999; 19: ) Miyata M, Takase S, Kobayashi H et al : Primary Sjögren s syndrome complicated by sarcoidosis. Intern Med 1998; 37: ) Singleton MA and Gros JC : Sialopathies in sarcoidosis. J Tenn Med Assoc 1970; 63:

過敏性肺臓炎との鑑別を要したサルコイドーシスの一例

過敏性肺臓炎との鑑別を要したサルコイドーシスの一例 1) 1) 1) 1) 1) 1) 1) 1) 2) 3) 要旨 76 2000 10 2 X 6 X キーワード : A Case of Sarcoidosis That Had Problems for Differential Diagnosis from Hypersensitivity Pneumonitis Shoko Matsui 1),