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Hereditary Haemorrhagic Telangiectasia (HHT) is a genetic disorder responsible for cutaneous or mucosal telangiectasia and arteriovenous malformations (AVMs). The most frequent locations are lung and brain. In contrast, orbital AVMs are very rare. We describe a case of symptomatic orbital arteriovenous malformation due to spontaneous thrombosis. A 65-year-old woman was referred for chronic right eye proptosis associated with dilation of conjunctival vessels with a jellyfish pattern. Right visual acuity was 20/40 and intraocular pressure was 40 mmHg. Personal and familial history of recurrent epistaxis, associated with multiple telangiectasia within lips and palate, led to the diagnosis of HHT. Magnetic resonance imaging (MRI) completed with cerebral angiography found a giant and occluded AVM within the right orbit. Other AVMs were also found in brain and chest, confirming the diagnosis. Antiglaucomatous eyedrops were added to reduce intraocular pressure and a steroid therapy was begun. Two months later, visual acuity decreased in the right eye, due to a central retinal vein thrombosis. In conclusion, Most brain or pulmonary AVM can be treated by embolization. By contrast, this treatment in case of orbital location can lead to central retinal artery and/or central retinal vein occlusion, which may also appear as a spontaneous complication of the orbital AVM. Therapeutic management of orbital AVM is thus not standardized, and the balance between spontaneous and iatrogenic risk of visual loss has to be taken into account.

Purpose To report the case of a patient with a corneal neurotrophic ulcer, resistant to the usual therapies, who has been treated with a new matrix therapy agent (RGTA). CACICOL 20® is a new ophthalmic device, derived from RGTA based matrix therapy (large biopolymers engineered to replace heparan sulfates). Methods We report the case of a monophtalm and deaf‐mute 48 years old woman, who presented with a central neurotrophic corneal ulcer, not responding to the first line therapies, including high dose lubricant eye drops and amniotic membrane graft. To avoid the profound confinement induced by tarsoraphy, a compassional treatment with RGTA CACICOL 20® has been proposed. CACICOL 20® was instilled once every other day for 2 weeks. Biomicroscopic pictures were recorded to follow the cicatrization time course. Results Epithelialization was obtained within 2 weeks with a perfect tolerance. The regimen of instillation was incidently doubled, causing a recurrence, which healed within 2 weeks after the correct regimen was reintroduced. Conclusion This case report is consistent with a strong effect of CACICOL 20® in the treatment of resistant neurotrophic corneal ulcers. The optimal posology and the indications needs to be refined.

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