ABSTRACT
BACKGROUND: Chorioretinal fold (CFs) is a rare condition resulting from undulations in the choriocapillaris, Bruch's membrane, retinal pigment epithelium and occasionally neurosensory retina. It can be idiopathic or due to different etiologies. The use of spectral-domain optical coherence tomography (SD-OCT) has increased the diagnosis of CFs and helped in differentiation from other etiologies. Recently, optical coherence tomography angiography (OCT-A) emerged as a non-invasive imaging technique allowing visualization of the individual layers of microvasculature of the retina and the choroid by comparing consecutive B-scans. We described a rare case of pleomorphic adenoma of the lacrimal gland (PALG) causing hyperopic shift and CFs with the new OCT-A technology, getting deeper insight into vascular changes of this disease. CASE PRESENTATION: A 40-year-old Asian man experienced progressive blurred vision in his right eye over 6 months. The patient's initial axial lengths were 25.55 mm in the right eye and 28.13 mm in the left eye. Fundus examination in the right eye revealed oblique CFs as well as the SD-OCT displayed. Magnetic resonance imaging showed intraconal mass extended from superior temporal side of the right orbit. The patient then received tumor removal surgery through lateral orbitotomy and histopathology confirmed a pleomorphic adenoma of the orbit. The patient had regular follow-up for 1 year. His best corrected visual acuity markedly improved from 20/50 to 20/20 with nearly stationary AXL. We performed OCT-A at one year after the surgery, which showed early visualization of deep choroidal vessels. The scleral remodeling due to mass effect of retrobulbar tumor also caused displacement of the deep large choroidal vessels over the superior macular area even after tumor removal. CONCLUSIONS: We reported a rare case of PALG with hyperopic shift and CFs as initial presentation. Surgical removal of the tumor partially resolved the CFs and contributes to impressive visual acuity recovery. The use of OCT-A provided a deeper insight to vascular architecture changes resulting from scleral remodeling after long-term tumor compression.