血管新生緑内障をきたした小児網膜中心静脈閉塞症

A case of neovascular glaucoma due to central retinal vein occlusion in a healthy child

要約　緒言：小児に網膜中心静脈閉塞症（CRVO）から血管新生緑内障を生じた稀な症例の報告。

症例：6歳，男児，双生児の弟。左眼視力低下と飛蚊症を訴え関西医科大学附属病院眼科を紹介され受診した。初診時左眼矯正視力は1.5，左眼圧は15mmHgで左眼底には視神経乳頭浮腫，網膜静脈の拡張蛇行と広範囲の網膜血管吻合，網膜出血，網膜静脈の白鞘形成，網膜のびまん性浮腫，黄斑の不完全な星芒斑を認めた。原因検索のため全身検査を行ったが異常はなかった。乳頭血管炎を疑いプレドニゾロン20mg/日内服を開始したが，網膜前出血が拡大したため1か月後にフルオレセイン蛍光眼底造影検査を施行したところ，網膜静脈と視神経乳頭部からの強い蛍光漏出と広範な網膜無灌流領域を認めた。出血はCRVOによる乳頭上新生血管が原因と考え，1.5か月後全身麻酔下に汎網膜光凝固とベバシズマブ硝子体内注射（IVB）を施行したが，網膜前出血がさらに拡大したため，5か月後に近畿大学病院にて硝子体手術とIVBを施行した。網膜復位2か月後に左眼圧52mmHgと上昇し血管新生緑内障を発症したため，再度光凝固追加とIVB，毛様体レーザー，トリアムシノロンテノン囊下注射を施行した。術後眼圧は下降し，現在眼圧の再上昇は認めないが，左眼視力は光覚弁となった。

結論：小児の乳頭血管炎を疑う場合でも，重篤な虚血型CRVOが発症しうることに注意し，早急な網膜虚血への対策が重要である。

Abstract　Introduction：A report of a rare case of neovascular glaucoma due to central retinal vein occlusion（CRVO）in a child.

Case report：A six-year-old male（the younger brother of twins）was consulted by our hospital with a complaint of visual loss and floater in the left eye. The corrected visual acuity of the left eye was 1.0, and intraocular pressure was 15 mmHg at the time of the initial examination. Fundus examination of the left eye showed optic disc edema, engorgement and tortuosity of retinal veins, many retinal arteriovenous shuntings, retinal hemorrhage, sheathing of retinal veins, diffuse edema of the retina, and incomplete starfigure in the macula. A systemic examination was performed to find the cause, but no abnormalities were found. Upon diagnosis of optic disc vasculitis, a corticosteroid was given systematically, but a the pre-retinal hemorrhage developed. Fluorescein angiography was performed one month later, which showed widespread non-perfusion areas, neovascularization in the optic disc（NVD）, and dye leakage from the optic disc and the retinal vein. We speculated that the cause of the aggravation would be due to NVD from CRVO. Six weeks later, we performed pan-retinal photocoagulation（PRP）, and intravitreal bevacizumab（IVB）was performed under general anesthesia. However the pre-retinal hemorrhage expanded further, so the patient underwent a vitrectomy and IVB at Kindai University Hospital five months later. Seven months later, the neovascular glaucoma progressed, so additional PRP, IVB, ciliary body laser, and sub-Tenon injection of triamcinolone were performed under general anesthesia. Postoperatively, intraocular pressure（IOP）decreased and there was no reincrease in IOP at the time this paper was written, but visual acuity of the left eye decreased to 30 cm finger counting.

Conclusion：It is important to note that severe ischemic CRVO due to optic disc vasculitis can occur even in a healthy child, and that immediate treatment must be taken to prevent the progression of retinal ischemia.