Unusual Presentation of Acute Hydroxychloroquine Retinopathy.

PURPOSE: To report a rare case of cystoid macular edema (CME) as a presentation of acute hydroxychloroquine-related retinal toxicity. OBSERVATIONS: A 37-year-old female patient visited our ophthalmology department in October 2019 complaining of bilateral blurred vision and metamorphopsia for 3 days. Best-corrected visual acuity (BCVA) was 6/6 in the right eye and 6/7.5 in the left eye under the Snellen E chart. Before presentation, she had taken hydroxychloroquine as a "reproduction-facilitating medication" prior to the in vitro fertilization (IVF) procedures with the daily dose of 200 mg for 1 week in March 2019 and 400 mg for 1 month in September 2019. She also took a combination of several herbal medicine including "Angelica sinensis" for 6 months in this period. On examination, typical signs of hydroxychloroquine maculopathy such as bilateral paracentral retinal pigment epithelium (RPE) change in blue autofluorescence and loss of the paracentral ellipsoid zone in optical coherence tomography ("flying saucer sign") were noted. CME was also found in fluorescein angiography. Her symptoms improved gradually after cessation of hydroxychloroquine and herb medicine without any further treatment. Resolution of bilateral CME was revealed at 16 weeks with final bilateral BCVA 6/6. CONCLUSIONS AND IMPORTANCE: Although rare, acute hydroxychloroquine maculopathy could occur in patients with concomitant usage of medications that could interfere with P450 enzymes system. Careful acquisition of drug history and serial ophthalmological examinations are advised in using hydroxychloroquine for disease management even for a short period of time.

PENTOSAN POLYSULFATE SODIUM-INDUCED PIGMENTARY MACULOPATHY WITH NONLEAKING CYSTOID MACULAR EDEMA SUCCESSFULLY TREATED WITH ANTI-VASCULAR ENDOTHELIAL GROWTH FACTOR THERAPY.

PURPOSE: To report a case of nonleaking cystoid macular edema (CME) associated with pentosan polysulfate sodium (PPS)-induced pigmentary maculopathy. METHODS: Multimodal imaging, including optical coherence tomography, fundus photography, autofluorescence, and fluorescein angiography, was used to substantiate our diagnosis, further characterize the cystoid macular edema showed by our patient and to monitor the response to treatment. RESULTS: A 59-year-old woman was referred for decreased visual acuity and bilateral macular edema. She had been treated for interstitial cystitis with PPS for 10 years. Multimodal imaging showed the characteristic features of PPS-induced pigmentary maculopathy. Moreover, fluorescein angiogram showed nonleaking cystoid macular edema in both eyes. She was treated successfully with intravitreal injections of bevacizumab. CONCLUSION: To our knowledge, this report is the first to demonstrate that PPS-associated cystoid macular edema can be nonleaking on fluorescein angiography and responds well to intravitreal anti-vascular endothelial growth factor injections.

[A rare case of cystoid maculopathy]. / Un cas rare de maculopathie cystoïde.

We describe the case of a healthy 67-year-old Caucasian female who developed a rare, reversible case of cystoid maculopathy without late leak on fluorescein angiogram. She presented initially with a 5-year history of bilateral, variable central scotomata. The visual acuities were 6/6, while foveal reflexes were abnormal. Fluorescein angiogram showed no leakage; however, optical coherence tomography (OCT) revealed cystic spaces in the fovea of both eyes. The patient had been taking niacin supplements averaging 100mg daily for 30 years. Niacin was discontinued, and 7 months later, her symptoms and OCT results improved. Although niacin-induced maculopathy is uncommon, the clinician should enquire about possible niacin supplementation in any patient with cystoid macular edema in the absence of late leakage on fluorescein angiography. We describe a new reason for niacin maculopathy due to chronic low dose niacin supplementation and a slower pattern of visual recovery than that previously suggested in the literature.

Irreversible ocular toxicity from single "challenge" dose of deferoxamine.

Deferoxamine is a chelating agent used in the treatment of transfusional iron overload and more recently in the diagnosis and treatment of increased aluminum body stores in chronic renal failure patients. High dose chronic and short-term treatment has been associated with ocular toxicity. We present a case of irreversible visual loss that occurred with a single small "challenge" dose of deferoxamine.