Terson’s syndrome with macular hole (MH) is rarely seen, and the mechanism of which is not clear. Here we report a case of Terson Syndrome with the inner limiting membrane (ILM) peeled off spontaneously associated with a rare finding: MH. Case presentation This report presents the case of a 36-year-old female patient with aneurysmal subarachnoid hemorrhage (SAH) and Terson syndrome in the right eye was admitted to our hospital with blurred vision in August 2018. Pas plana vitrectomy (PPV) was performed in the right eye, a hyaloid detachment and dyeing of the ILM with indocyanine green (ICG) was assisted. After removal of the vitreous hemorrhage (VH), a full-thickness MH was noted and we also noticed a particular aspect: the ILM was already peeled spontaneously. So we conducted gas tamponade, and face-down positioning after PPV. At two weeks follow-up, spectral domain optical coherence tomography (SD-OCT) confirmed that the MH had closed, while the thickness of nasal retina was 0.137 nm thicker than that in the temporal side. Her best corrected visual acuity (BCVA) was 0.15 in the right eye and 1.0 in the left eye. Conclusions MH is a rarely seen complication of Terson Syndrome. We conferred that the pathogenic mechanisms of this unusual MH may include stretching forces at the ILM-macular interface.