Introduction: Mycoplasma pneumoniae is known as a common cause of respiratory tract infections, especially in children. Regarding extrapulmonary manifestations, many dysfunctions have been linked to circulating IgM antibodies, including eye diseases and disorders. In this report, we aim to highlight the importance of considering Mycoplasma pneumoniae a potential etiological agent that can cause significant eye structures inflammation. Case presentation: We present a case of a 22-year-old male patient who arrived at the Emergency Department complaining of visual acuity decrement. Fundoscopic examination outlined a pale optic nerve, covered by pre-papillary infiltrates and peripheric inflammatory infiltrates, accompanied by signs of vasculitis. Investigations were performed and a multidisciplinary assessment was conducted. General antibiotic and antimycotic treatment and topical non-steroidal anti-inflammatory drops were administrated but his symptoms were aggravating, although it was continuously upgraded. Antibodies for Mycoplasma pneumoniae were determined with positive IgM and macrolide antibiotherapy was administrated, with favorable evolution. Conclusions: Mycoplasma pneumoniae should not be excluded as a possible cause of severe ocular inflammations, even in asymptomatic patients. The patient’s management should include multidisciplinary assessment for an easier diagnosis in cases of uncertainty.