Post-operative optical coherence tomography angiography features of chorioretinal folds resulting from pleomorphic adenoma of the lacrimal gland (PALG) of orbit- a case report

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.

Quantitative analysis of retinal and choroidal vasculature in patients with chorioretinal folds secondary to hyperopia

“…These results are contrary to what was previously reported in the literature, which consists of the presence of linear signal reductions in the DCP and CC slabs. 1,2,7 Nonetheless, our study differed from the previous reports in that it only includes patients with CRF secondary to hyperopia. Furthermore, we were able to demonstrate the presence of linear transversal signal attenuations in both DCP and CC layers in one eye of one of the patients with CRF (Figure 2(d) and 2(e)).…”

“…7 Chen et al presented a case of CRF secondary to a pleomorphic adenoma of the lacrimal gland of orbit, and described the presence of areas with reduction of the signal in the DCP and in the CC slabs. 2…”

“…7 Chen et al presented a case of CRF secondary to a pleomorphic adenoma of the lacrimal gland of orbit, and described the presence of areas with reduction of the signal in the DCP and in the CC slabs. 2 In our study, we planned to investigate vascular alterations secondary to CRF measuring quantitative parameters of OCTA, including VD of SCP, MCP, DCP, and CC, and FAZ area of SCP, MCP, and DCP. Our results showed a significant diminution of foveal VD in the SCP and a significant enlargement of FAZ area in the SCP.…”

“…Recently, optical coherence tomography angiography (OCTA) characteristics in patients with CRF have been reported and the authors described the presence of a linear signal reduction in the choriocapillaris slab in correspondence of the fold. 1,2,7 Nevertheless, there is a lack of information on quantitative parameters of OCTA in cases of CRF in the current literature.…”

“…Chorioretinal folds (CRF) are a relatively uncommon clinical sign resulting from undulations of the choroid, Bruch's membrane (BM), retinal pigment epithelium (RPE), and the overlying neurosensory retina. [1][2][3] Rarely, CRF may cause symptoms such as metamorphopsia, photopsia, or blurred vision. Thus, this clinical sign is usually discovered during routine ophthalmological examinations.…”