シェーグレン症候群とHTLV-1感染が関与したと考えられる気管支肺病変の1例

A Case of Bronchopneumopathy Complicated with Sjögren's Syndrome in Human T-cell Lymphotropic Virus Type 1 Carrier

要旨　症例は44歳女性．肺炎の精査加療目的で近医より紹介入院した．抗菌薬の投与を行ったところ，一部の陰影を残し肺野陰影の改善を認めた．残存した肺野病変の精査目的で胸腔鏡下肺生検を施行し，リンパ球や形質細胞の浸潤を伴ったnonspecific interstitial pneumoniaパターンやorganizing pneumoniaパターンの間質性肺炎と診断した．胸腔鏡下肺生検の結果およびシルマーテストが陽性であること，唾液腺の生検でリンパ球の浸潤を認めたことより，シェーグレン症候群と診断した．また血液検査で，HTLV-1キャリアであることが判明した．残存した肺野病変に対して，クラリスロマイシンの内服を開始し経過観察を行ったところ，陰影の改善を認めた．シェーグレン症候群とHTLV-1感染が関与したと考えられる気管支肺病変の報告は少なく，貴重な症例と考えられた．

　A 44-year-old woman was admitted to our hospital because of high fever. A chest computed tomography(CT)showed a thickened bronchial wall, a dilated bronchial lumen, centrilobular nodules, and infiltrative shadows in the bilateral lower lung fields. On suspicion of bronchopneumonia, cefozopran was administered. Her symptoms were improved and infiltrative shadows were diminished, but some of centrilobular nodules remained unchanged. In order to confirm her precise diagnosis, video assisted thoracoscopic biopsy was performed and histopathological examination revealed non-specific interstitial pneumonia and organizing pneumonia patterns. She had suffered from dry mouth and had lost same of teeth. From the results of Schirmer's test, salivary gland biopsy was performed, and we concluded that this was a case of Sjögren's syndrome. Moreover, she was also found to be a human T-cell lymphotropic virus type 1(HTLV-1)carrier. We finally diagnosed bronchopneumopathy complicated with Sjögren's syndrome in a HTLV-1 carrier. Clarithromycin was administered for bronchopneumopathy, and chest CT after 1-year follow-up revealed that residual shadows had improved. This is a rare case of Sjögren's syndrome in a HTLV-1 carrier manifesting bronchopneumopathy.