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A. Balmer, M. C. Gaillard, F. L. Munier; Potential Role of Intravitreal Ranibizumab (lucentis®) in the Management of Stage 4 Coats Disease: Report of Two Cases. Invest. Ophthalmol. Vis. Sci. 2007;48(13):5771.
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Coats disease is a rare pediatric condition characterized by a developmental abnormality of the retinal vessels (telangectesia) leading to an exsudative retinopathy. The prognosis of eye conservation is directly related to the disease stage, with the most advanced one (total retinal detachment and neovascular glaucoma) being most frequently doomed to enucleation.
In this study we report the preliminary results of 2 consecutive cases of 10 months (case A) and 14 months of age (case B) with total retinal detachment, virtual vitreous cavity, rubeosis iridis and neovascular glaucoma, diagnosed at Jules Gonin Eye Hospital between June and September 2006. After ethical approval (Swissmedic) and informed consent from the parents, compassionate use of ranibizumab was obtained and the 2 patients were treated by a combination of a single intravitreal ranibizumab injection (0.3mg and 0.5mg respectively), drainage of the subretinal fluid and iterative argon laser photocoagulations.
Follow-up was 4 and 2 months respectively. Neovascular glaucoma and rubeosis iridis totally regressed within a week in both patients, allowing for a better pupil dilation and hence bringing peripheral telangiectasia to more extensive laser photocoagulation. Complete reattachment of the retina was observed within 7 weeks in case A and 6 weeks in case B. There were no acute or late side effects observed.
The spectacular rescue achieved by mean of intravitreal ranibizumab can be explained by the pharmacologic effect of ranibizumab on both the permeability of the abnormal retinal vascular bed as well as on the anterior chamber neovascularisation, which in turn led to a better visualization of telangiectasia for the purpose of photocoagulation. The role of anti-VEGF therapy in Coats disease needs to be further investigated.
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