RESUMEN
Fibrous dysplasia (FD) is a progressive and benign osteodystrophic disease where cranial bones are most commonly affected. In this case report, we present a 27-year-old patient with previous diagnosis of FD who was referred to our clinic with sudden loss of visual acuity and color discrimination. Examination of the right eye was normal, whereas visual acuity on the left eye was 6/9 and color vision (CV) with Ishihara test plates was 9/12. The visual field (VF) demonstrated a peripheral concentric defect on the left eye. As visual acuity in the left eye decreased to 6/30 and computed tomography imaging of the brain and orbit showed optic nerve compression by immature bony structures, optic nerve decompression was recommended with the diagnosis of compressive optic neuropathy. Endoscopic transnasal orbital and optic canal decompression was performed. At the postoperative course, visual acuity on the left eye turned to 6/6, CV was 12/12, and VF improved markedly. In subjects with craniofacial FD, a multidisciplinary approach is important. If there is evidence of compressive optic neuropathy, surgery should be performed.
RESUMEN
PURPOSE: To compare vascular microcirculation changes of the optic nerve head (ONH) and to evaluate the peripapillary retinal nerve fiber layer (RNFL) in the patients with unilateral pseudoexfoliation syndrome (PEXS), other eyes of these patients without PEXS and healthy control eyes using optical coherence tomography angiography (OCTA) and spectral domain optical coherence tomography (SD-OCT). METHODS: This cross-sectional study enrolled 29 pseudoexfoliative (PEX) and 29 fellow eyes of patients with unilateral PEXS, and 28 healthy eyes as controls. According to inclusion criteria the study group with asymmetric PEXS were normotensive and no glaucoma diagnosis. The vascularity of optic disc was evaluated with OCTA and peripapillary retinal nerve fiber layer with SD-OCT. ONH 4.5 × 4.5 mm OCTA gave us perfusion density (PD) and flow index (FI). Differences between the groups according to continuous variables were determined by independent samples t test. A p value less than 0.05 was considered as significant. RESULTS: Twenty-nine patients with the mean age of 66.21 ± 7.55 (49-79) years was compared with 28 individuals in control group with mean age of 66.79 ± 4.43 (60-75) years. There was no difference regarding the age (p = 0.725). Female and male distribution in two groups were same (p = 0.219). Intraocular pressure (IOP) in PEXS group was measured as 16.17 ± 4.58 (10-21) mmHg, however IOP in the fellow eye was measured as 14.79 ± 3.35 (11-19) mmHg (p = 0.064) and control group was measured as 12.53 ± 1.66 (10-17) mmHg (p = 0.000). In the group with PEXS, the superior FI was found to be lower 0.39 ± 0.06 (0.26-0.45) (p = 0.008) than control group 0.42 ± 0.21 (0.36-0.45), and temporal FI in PEXS eyes was measured 0.42 ± 0.06 (0.32-0.52), which was significantly lower than control group (p = 0.022). Nasal FI was the parameter which was found significantly different from the PEXS free fellow eyes. The nasal FI value of eyes with PEX was 0.40 ± 0.05 (0.30-0.46), while the same value in PEX free fellow eyes was 0.42 ± 0.04 (0.33-0.47) (p = 0.029). CONCLUSIONS: ONH evaluation with OCTA especially in the nasal segment is important for early diagnosis. OCTA can be a new method in follow-up and early diagnosis of patients with asymmetric PEXS to control ONH microcirculation and to evaluate early glaucomatose changes in both eyes.