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.

Thiamine-deficient optic neuropathy associated with Wernicke's encephalopathy in patients with chronic diarrhea.

The deficiency of thiamine manifesting as Wernicke's encephalopathy (WE) and concurrent optic neuropathy is rare. Herein, we report the case of a 29-year-old patient who suffered from bilateral sudden blindness and a disturbance of consciousness after 2 months of chronic diarrhea and minimal food intake. In addition, bilateral abducens nerve palsy with multidirectional nystagmus and no light perception in both eyes were noted. An ophthalmoscopic examination revealed bilateral disc edema with peripapillary flame-shaped hemorrhages. Although the results of analyzing the composition of cerebrospinal fluid showed that they are within normal limits, magnetic resonance imaging (MRI) revealed bilateral hyperintensity over the mammillary body, dorsal medial thalamus, and periaqueductal gray matter. As we suspected thiamine deficiency-induced WE, a high dose of intravenous thiamine was prescribed. After the administration of thiamine, both visual acuity and visual field rapidly improved with the simultaneous recovery of consciousness. This case indicates that, although rare, thiamine deficiency with WE may still occur in patients with chronic diarrhea in Taiwan. Thiamine deficiency should be considered in the differential diagnosis for patients who encounter sudden visual loss after prolonged periods of poor food intake and poor vitamin supplementation.