A Case of Sarcoidosis Manifesting Liver Involvement after Disappearance of Muscle Lesion Hirohisa Ichikawa1), Mikio Kataoka2), Yoshiko Ogata1), Masaka

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A Case of Sarcoidosis Manifesting Liver Involvement after Disappearance of Muscle Lesion Hirohisa Ichikawa1), Mikio Kataoka2), Yoshiko Ogata1), Masakazu Ohmori1), Junichi Hiramatsu1), Yasusi Tanimoto1), Arihiko Kanehiro1), Yasunari Nakata2), Mitsune Tanimoto1) ABSTRACT z We report a case of a 42-year-old man who had bilateral hilar lymphadenopathy and small nodular shadows in the left upper lung field detected by a health check-up at the age of 35. He was diagnosed as having sarcoidosis by TBLB. He had a nodule on his right thigh and was diagnosed as having muscular sarcoidosis by muscle biopsy. We carefully followed-up the disease course without any treatment, and the tumor finally disappeared. At the age of 42, he suffered from back pain and went to a hospital. Because his abdominal CT revealed an irregular surface of the liver and multiple hypodense lesions in the liver and spleen, he was admitted to our hospital for further examinations. A laparoscopy revealed white plaque covering the liver. Biopsy specimens showed non-caseating granulomas. He was diagnosed as having liver sarcoidosis and treated with prednisolone (40mg/day). He died from a sudden accident after this. Here, we report a valuable case in which nodular muscular sarcoidosis and hepatic sarcoi dosis were seen at different times. keywords; Nodular muscular sarcoidosis, Hepatic sarcoidosis, Laparoscopic liver biopsy [JJSOG 2004; 24: 71-76] TEL: 086-235-7227 FAX: 086-232-8226 E-mail: hirohisa@cc.okayama-u.ac.jp Okayama University Medical School 1) Hematology, Oncology and Respiratory Medicine 2) Faculty of Health Sciences

Table 1. Data on admission

Figure 1a. Chest X-ray on admission showing small nodular shad ows in left upper lung field and bilateral hilar lymphaden opathy Figure 1b. 1c. Chest CT on admission showing small nodular shadows in lung fields and bilateral hilar lymphadenopathy Figure 2a. Gd-enhanced T1 weighted MR-image of right thigh in 1996, showing flat tumor with Gd enhancement (arrows) Figure 2b. T1 weighted MR-image of right thigh in 2003, the tumor disappeared

Figure 3a. Abdominal CT in 1996, showing fatty liver Figure 3b. Abdominal CT in 2003, showing irregular surface of liver, mild splenomegaly and multiple small hypodense hepatic and splenic nodules Figure 4a. T1 weighted MR-images on admission, showing hypointense reticular and small nodular shadows in liver Figure 4b. T2 weighted MR-images on admission, showing hyper intense reticular shadows in liver and small hypointense nodular shadows in spleen Figure 5a. Laparoscopy revealed white plaque covering liver Figure 5b. A part of gall bladder with sarcoid nodule adhering (arrow)

Figure 6. Histological findings of a liver biopsy specimen, showing non-caseating epithelioid cell granulomas with fibrotic tissues. (HE, ~100) Figure 7. Clinical course of this patient

1) Silverstein A,Siltzbach LE: Muscle involvement in sarcoido sis. Asymptomatic myositis and myopathy. Arch Neurol 1969; 21: 235-241. 4) Iwai K, Takemura T, Kitaichi M, et al: Pathological studies on sarcoidosis autopsy. II Early change, mode of progression and death pattern. Acta Pathol Jpn. 1993; 43: 377-385. 5) Hunninghake GW, Costabel U, Ando M, et al: ATS/ERS/ WASOG statement on sarcoidosis. Sarcoidosis Vasc Diffuse Lung Dis 1999; 16: 149-173. 7) Warshauer DM, Lee JK: Imaging manifestations of abdomi nal sarcoidosis. Am J Roentgenol 2004; 182: 15-28. 8) Scott GC, Berman JM, Higgins Jr JL: CT patterns of nodular hepatic and splenic sarcoidosis: A review of the literature. J Comput Assist Tomogr 1997; 21: 369-372. 9) Folz SJ, Johnson CD, Swensen SJ: Abdominal manifesta tions of sarcoidosis 1995; 19: 573-579. in CT studies. J Comput Assist Tomogr 10) Wallace SL, Lattes R, Malia JP, et al: Muscle involve-ment in Boeck's sarcoid. Ann Int Med 1958; 48: 497-511. 11) Hinterbuchner CN, Hinterbuchner LP: Myopathic syndrome in muscular sarcoidosis. Brain. 1964: 87: 355-366. 13) Warshauer DM, Molina PL, Hamman SM, et al: Nodular sar coidosis of the liver and spleen: analysis of 32 cases. ology 1995; 195: 757-762. Radi 15) Lynch JP III, Sharma OP, Saughman RP: Extrapulmonary sarcoidosis. Semin Respir Infect. 1998; 13: 229-254. 17) Kataoka M, Nakata Y, Hiramatsu J, et al: Hepaticand splenic sarcoidosis evaluated by multiple imaging modalities.lnter Med 1998; 37: 449-453.

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