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Trakya Ü. Tıp Fakültesi Göz Hastalıkları A.D., Edirne. Facioscapulohumeral Muscular Dystrophy (FSHD),is a progressive muscular disorder. Typical features include wasting of the facial or shoulder girdle muscles. Facial weakness affects the eye closure (orbicularis oculi). Progressive hearing loss and retinal vasculopathy with capillary telangiectasis, microaneurysms and capillary closure has been reported. Ophthalmoscopic examination is not enough to establish the retinal pathologic changes, due to capillary occlusion. Since visual loss is possible, FFA is indicated in all patients with facioscapulohumeral syndrome. In this report, facial and retinal changes are demonstrated in a case with familial FSHD without hearing loss. Keywords : Fasioscapulohumeral Muscular Dystrophy,Lagophthalmy, Telangiectasy