サルコイドーシスの経過観察中に頸部リンパ節生検で診断したT細胞リンパ腫を合併した1例

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1 T 1 サルコイドーシスの経過観察中に頸部リンパ節生検で診断した T 細胞リンパ腫を合併した 1 例要旨 67X 3 6T 日サ会誌 2005;25:53-59 キーワード : T A Case of Sarcoidosis Who Developed T-cell Lymphoma Diagnosed by Cervical Lymph Node Biopsy Kazuhiko Terakawa 1), Takashi Kawaguchi 1), Satoshi Shiraishi 1), Yoshikazu Hasegawa 2), Hirofumi Katayama 2), Mituhiro Sumitani 2), Takeshi Inoue 3), Takamune Sugiura 4) ABSTRACT We experienced a case of sarcoidosis who developed T-cell lymphoma diagnosed by cervical lymph node biopsy. This 67 y/o male was followed due to gastric cancer operation and ossification of posterior longitudinal ligament. He was found to have abnormal chest X-ray. Transbronchial lung biopsy revealed sarcoidosis. He noticed cervical lymph node swelling. Cervical lymph node biopsy revealed sarcoidosis. When he again noticed cervical lymph node swelling another lymph node biopsy revealed T-cell lymphoma. We reported this rare case of sarcoidosis accompanied with T-cell lymphoma. [JJSOG 2005;25:53-59] keywords ; Sarcoidosis, Lymph node swelling, T-cell lymphoma 1) 大阪市立北市民病院呼吸器科 2) 大阪市立総合医療センター呼吸器内科 3) 大阪市立総合医療センター病理部 4) りんくう総合医療センター市立泉佐野病院呼吸器センター著者連絡先 : 寺川和彦大阪市此花区西九条 5 丁目 4-8 大阪市立北市民病院呼吸器科 TEL: FAX: [email protected] 1) Department of Respirology Osaka City Kita Hospital 2) Department of Respiratory Medicine Osaka City General Hospital 3) Department of Pathology Osaka City General Hospital 4) Department of Respirology Rinku General Hospital 53

2 日サ会誌 2005, 25(1) はじめに 2 T Brincker 1 Sarcoidosis-lymphoma syndrome 5.5 T 症例提示症例 :67 主訴 : 既往歴 :50 C 喫煙歴 :2040 家族歴 : 現病歴 : BHL TBLB TBLB BAL CT peripheral T lymphoepithelioid cell variant 162cm 52kg mmHg 75 2cm Table 1 ACE 43.2mg dl HCV 8mm 201 Tl RI Urinalysis Table 1. Laboratory Data on First Visit ph 6 protein (-) glucose (-) occult blood ( ) CBC WBC 4300/ RBC / Hb 11.1 g/dl Ht 34 % Plt / ( Neut 51 Lymp 35 Mono 7 Eos 3 Bas 1 ) Blood chemistry AST 25 ALT 19 Alp 316 LDH 234 LAP 54 ChE 220 GTP 44 Amylase 123 T-Bil 0.5 TP 7.2 g/dl ( Alb ) BUN 13 mg/dl Cre 0.8 mg/dl UA 5 mg/dl Na 140 K 4 Cl 106 Ca 9mg/dl T Chol 183 mg/dl TG 107 mg/dl BS 103mg/dl Serum CRP 0.05 mg/dl IgG 1619 IgA 455 IgM 604 IgE 145 C3 67 C4 17 Mycoplasma <40 RF 1 IU/ml ANA 20 ACE 43.2 IU/l HCV(+) Tumor Maker CEA 3 ng/dl CA U/ml CYFRA 1 ng/ml ProGRP 8.5 pg/ml PPD:8mm 8mm( negative ) Arterial Blodd Gas Analyisis ( room air ) ph 7.41 Pco2 38 Po2 87 Hco3 24 BE 0.1 Sao2 95 Pulmonary Function Data VC 2760 ml ( 83.6 % ) FVC 2760 ml ( 83,6% ) FEV ml FEV1.0% 83% FRC 2830 ml ( 84.1 % ) RV/TLC 63.2%DLco 12.4 ml/m/mmhg ( 70.8 % ) DLco/VA 3.8 ( 85 % ) 臨床経過 Figure1 CT Figure Figure 2a RI TBLB1997 9Figure 2b Figure 2c RI Figure 2d RI CT Figure 4 54

T 1 T Chest X-ray HRCT ( July 1997 ) July 25 1997 December 2 2003

PA chest radiograph and CT showed mild bilateral hilar lymph node

mediastinal lymph node uptake (By courtesy of the hospital previously

2b: 67 Ga Scintigram on September 8, 1997 revealed decrease og uptake

3 T 1 T Chest X-ray HRCT ( July 1997 ) July December Figure 1. PA chest radiograph and CT showed mild bilateral hilar lymph node swelling and diffuse shadows of the lung. Figure Ga Scintigram of the patient. 2a: 67 Ga Scintigram on April 10, 1997 revealed cervical and mediastinal lymph node uptake (By courtesy of the hospital previously in charge of the patient). 2b: 67 Ga Scintigram on September 8, 1997 revealed decrease og uptake (first visit). 2c: 67 Ga Scintigram on March 18, 2001 revealed submandibular uptake (first cervical lymph node swelling). 2d: 67 Ga Scintigram on August 22, 2003 revealed cervical,mediastinal and abdominal lymph node uptake (second cervical lymph node swelling). 55

日サ会誌 2005, 25(1) TBLB BAL Figure 3Figure 3a TBLB

4 日サ会誌 2005, 25(1) TBLB BAL Figure 3Figure 3a TBLB microgranuloma Figure 3b TBLBFigure 3c BAL Figure 3d Figure 3. TBLB, BAL and pathological findings of cervical lymph node revealed sarcoidosis. 3a:TBLB,3b:TBLB,3c:cervical lymph node biopsy CT Figure4 CT Figure5 CTFigure5 Figure6 CD3 T T T 5 56

Submandibular lymph node swelling Supraclavicular lymph node swelling

5 T 1 Lymph node swelling Lymph node swelling Figure 4. Multiple abdominal lymph node swelling on CT. Submandibular lymph node swelling Supraclavicular lymph node swelling Figure 5. Multiple cervical lymph node swellings and ossification of posterior longitudinal ligament on CT. (OPLL: 後縦靭帯骨化症 ) 57

6 日サ会誌 2005, 25(1) Figure 6. Histological specimen of the cervical lymph node revealed peripheral T-cell lymphoma (HE and immunohistogical stainings). 考察 Brincker 1 sarcoidosis-lymphoma syndorome T T 1997 T 1 HTLV-I T sil2r

7 T 1 結論 T IL 引用文献 1) Brincker H: The sarcoidosis-lymphoma syndorome. Br J Cancer 1986; 54: ) 立花暉夫, 竹中雅彦, 井上義一他 : 大阪地区サルコイドーシス症例の合併症に関する検討. 日サ会誌 2004;24: ) Gelfand JM, Wasik MA and Vittorio C et al: Progressive epidermotropic CD8+/CD4- primary cutaneous CD30+ lymphproliferative disorder in a patient with sarcoidosis. J Am Acad Dermatol 2004; 51: ) 仁田正和 :sarcoidosis-lymphoma syndrome. 日本臨牀別冊領域別症候群血液症候群 III 造血器悪性腫瘍と類縁疾患非 Hodgkinリンパ腫.1998; ) Sawhney M, Sharma YK and Gera V et al: Ichythyosiform sarcoidosis following chemotherapy of Hodgkin s Disease. Indian J Derma Venerelogy and Leprology 2003; 69: ) Sacks EL, Donaldson SS and Gordon J, et al: Epithelioid granulomas associated with Hodkin s disease: clinical correlation in 55 previously treated patients. Cancer. 1978; 42: ) Yajima A, Kawada A, Aragane Y et al: Detection of HTLV-I proviral DNA in sarcoidosis Dermatology 2001; 203: ) Lower EE, Hawking H, Baughman R: Breast disease in sarcoidosis. Sarcoidosis Vasc Diffuse Lung Dis 2001; 18: ) Brown JR, Skarin AT. Clinical mimics of lymphoma Oncologist. 2004; 9: ) 中山智子, 峰須賀久喜, 古市祥子他 : 縦隔腫瘍と鑑別を要し胸腔鏡下生検にて診断し得たサルコイドーシスの 1 例. 日呼吸会誌 2000;38:

日本呼吸器学会雑誌第44巻第10号

日本呼吸器学会雑誌第44巻第10号 β Table1 Laboratoryfindingsonthefirstvisit Hematology WBC Neut. Lyn. Mon. Eos. Bas. RBC Hb Ht Plate 5,650 /μl 79.2 % 14.9 % 3.9 % 1.8 % 0.2 % 418 10 4 /μl 12.5g/dl 38.4 % 21.2 10 4 /μl Serology CRP IgG