To present retinal pigment epithelium (RPE) aperture related to an avascular pigment epithelium detachment (PED) secondary to acute central serous chorioretinopathy (CSC). Case report. A 47-year-old man diagnosed as acute CSC presented with RPE aperture in the superonasal area of the macula in his left eye during follow-up. At 2-week follow-up, his decimal best-corrected visual acuity (BCVA) was improved from 0.08 to 0.6 and subretinal fluid was partially absorbed. However, the near-infrared reflectance demonstrated a round mild hyperreflective lesion on the superonasal area of the macula. On spectral-domain optical coherence tomography (SD-OCT), RPE band of the round lesion discontinued but RPE fractured edges without shrinkage and curling. Fundus autofluorescence (FAF) showed RPE aperture appeared as round hypoautofluorescence and hyperautofluorescence outlined its borderline. OCT angiography demonstrated that no evidence of neovascularization within the sub-RPE space. En Face OCT confirmed that the RPE aperture developed at the edge of the PED lesion. At 8-month follow-up, his decimal BCVA was improved to 1.0 and SD-OCT demonstrated spontaneous resolution of subretinal fluid and restoration of RPE structure, with complete flattening of PED. However, FAF revealed hypoautofluorescence mingled with slight hyperautofluorescence within the lesion. To the best of our knowledge, this is the first report of an RPE aperture secondary to acute CSC. Our case indicated another novel possible pathological mechanism that in the relatively healthy RPE, increased hydrostatic pressure simply itself could contribute to RPE aperture.