胞状の滲出性網膜剝離が自然軽快したCoats病の1例

Spontaneous regression of bullous retinal detachment in a case of Coats disease

要約　目的：網膜全剝離が自然軽快したCoats病の1例報告。

症例：3歳9か月の男児。瞬目の増加で近医を受診し，右眼の網膜剝離を指摘され，名古屋市立大学病院を受診した。視力は右光覚弁なし，左1.0であった。右眼に胞状の網膜全剝離，網膜血管異常および網膜下滲出斑を認め，蛍光眼底造影検査で蛍光漏出および無灌流領域を認めた。超音波検査，CTおよびMRIでは充実性病変や石灰化はなく，Coats病と診断した。網膜電図は平坦で，光覚もないため，経過観察とした。初診から6か月後，徐々に末梢血管異常が改善し，滲出斑も減少してきた。それに伴い網膜剝離も改善したが，視力は光覚弁なしのままであった。

結論：本症例は経過中に末梢血管異常が改善し，血管透過性が減少したことにより滲出性網膜剝離が改善したと考える。

Abstract　Purpose: To report a case of Coats disease in which spontaneous regression of exudative total retinal detachment.

Case: A 3 years and 9 months old boy presented at local hospital after his mother noticed his remarkable increase of blinking. He was diagnosed retinal detachment in this right eye and was referred to Nagoya City University Hospital. Slit-lamp and fundus examination showed bullous retinal detachment just behind the lens. Abnormal retinal vessels were present, as well as the deposition of subretinal hard exudate. Marked fluorescein leakage from abnormal retinal vessels and non-perfusion area was revealed by fluorescein angiography. No other findings suggesting a mass legion or calcification was detected by ultrasound, CT and MRI. He was diagnosed as Coats disease complicated with total retinal detachment in his right eye. Due to no light perception and non-recordable electroretinogram in his right eye, no surgical treatment was indicated. Six months after the initial diagnosis, follow-up examination showed the retinal detachment to have spontaneously regressed as well as decreased vessel abnormality and subretinal hard exudates. Light perception was still absent.

Conclusions: The spontaneous regression of exudative retinal detachment may have been caused by decreased hyperpermeability from abnormal retinal vessels.