• Shuhei Hosoda, Yoichi Sakurada, Taiyo Shijo, and Kenji Kashiwagi.
• Department of Ophthalmology, Faculty of Medicine, University of Yamanashi, Yamanashi, Japan.
• Medicine (Baltimore). 2022 Dec 2; 101 (48): e32032e32032.

RationaleDrusen are precursor lesions to advanced age-related macular degeneration. Although cuticular drusen are located between the retinal pigment epithelium and Bruch's membrane, as are conventional drusen, they possess unique characteristics that are distinct from those of conventional drusen on clinical presentations. Central serous chorioretinopathy (CSC) is a rare complication in eyes with cuticular drusen.Patient ConcernA 58-years-old man was referred to our institute for the treatment of persistent subretinal fluid (SRF) in both eyes. Spectral-domain optical coherence tomography revealed focal SRF that did not involve the central macula of the right eye and SRF in the central macula of the left eye. Fluorescein angiography exhibited focal leakage corresponding to SRF and hyperfluorescence resembling a "stars in the sky" appearance in both eyes. On initial presentation, the best-corrected visual acuity values were 1.2 and 0.9 in the right and left eye decimal formats, respectively.DiagnosisCuticular drusen presenting with CSC in both eyes.InterventionsNo treatment was administered for CSC in the right eye, whereas photodynamic therapy was administered for CSC in the left eye.OutcomesAt the 6-month visit, extrafoveal SRF persisted in the right eye and resolved in the left eye. Best-corrected visual acuity improved from 0.9 to 1.2 in the decimal format in the left eye.LessonsAlthough cuticular drusen presenting with CSC are rare, physicians should be aware of the possibility of CSC development in eyes with cuticular drusen.Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc.

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