Introduction: Fibrous dysplasia (FD) of bone is a rare congenital disease characterized by a focal proliferation of fibrous tissue in the bone marrow, leading to osteolytic lesions, deformities and fractures. Purpose: To present a case of exophthalmos as the one clinical manifestation of fibrous dysplasia. Case: A 9-year-old girl with exophthalmos of the left eye was underwent the ophthalmological examination and computed tomography. The both eyes had a visual acuity of 20/20. The visual field was normal. On computed scan, the patient had a characteristic, pageotic appearance. Conclusion: Exophthalmos may be the only one clinical finding in fibrous dysplasia and should be a concern because of the possibility of visual impairment when the disease is progressive. The proper diagnosis may be necessary for the choice medical or surgical treatment and may be effective in preventing visual loss in FD patients. Long-term radiological and pediatric monitoring is essential. Therefore the ophthalmologist plays an important role in the early diagnosis of fibrous dysplasia
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