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Abstract Background Peripapillary intrachoroidal cavitation (PICC), a cavernous change in the peripapillary choroid, may present with macular lesions. Here, we present a rare case of PICC with outer lamellar macular hole and macular retinoschisis. Case presentation A 69-year-old man presented with metamorphopsia in the left eye. Fundus examination revealed macular retinoschisis and PICCs. Pars plana vitrectomy with fovea sparing internal limiting membrane peeling (FSIP) was performed. Three weeks postoperatively, the patient developed rhegmatogenous retinal detachment and underwent a second vitrectomy. The macular retinoschisis resolved without macular hole formation and the patient’s visual acuity improved. There were no recurrent macular lesions over the three years of postoperative follow-up. Postoperative spectral-domain optical coherence tomography scan revealed a communication between the PICC and the vitreous cavity. Conclusions This case shed light on understanding development of PICC in non-highly myopic eyes and a rare complication of macular retinoschisis. Vitrectomy with FSIP may effectively resolve macular retinoschisis.