難治性特発性眼窩炎症に対するシクロホスファミドの使用経験

Systemic cyclophosphamide for recalcitrant idiopathic orbital inflammation

要約　目的：ステロイド薬の全身投与で再燃を繰り返した特発性眼窩炎症に対し，シクロホスファミドで加療した2症例の報告。

症例：1例は38歳女性で，特発性眼窩炎症が左眼周囲に発症し，ステロイド薬の全身投与で再燃を繰り返し，眼球癆になった。5年後に右眼窩内に腫瘤が生じ，ステロイド薬に抵抗したためシクロホスファミドを経口投与した。腫瘤は速やかに消失し，1年後の現在まで再発はない。他の1例は23歳男性で，右視神経周囲に特発性眼窩炎症が発症した。ステロイド薬の大量投与で加療したが，6か月間に再燃を繰り返した。シクロホスファミド投与で腫瘤は速やかに消失し，9か月後の現在まで再発はない。

結論：ステロイド薬の全身投与で再発を繰り返す特発性眼窩炎症に対し，シクロホスファミドの経口投与が奏効した。

Abstract　Purpase: To report 2 cases of idiopathic orbital inflammation that responded to peroral cyclophosphamide after resisting systemic steroid therapy.

Cases: One was a 38-year-old woman with severe scleritis in her left eye that resulted in phitisis. An inflammatory mass appeared in the right lacrimal sac area 5 years later. The lesion did not respond to pulsed steroid therapy. It rapidly resolved after peroral cyclophosphamide therapy. There has been no recurrence for one year until present. The second case was a 23-year-old male who showed inflammatory masses around the right optic nerve. The lesions recurred repeatedly after reduction of dosis of steroid. The lesions resolved after peroral cyclophosphamide. There has been no recurrence for 9 months until present.

Conclusion: Peroral cyclophosphamide induced lasting resolution in 2 cases of idiopathic orbital inflammation that initially failed to respond to systemic steroid therapy.