SUMMARY: Dysthyroid optic neuropathy (DON) is the most serious complication associated with Graves' orbitopathy (GO). The primary treatment approach for DON is high-dose intravenous methylprednisolone infusion and decompression surgery. However, not all patients are suitable candidates for or can tolerate these two treatment options. Here, we present a patient diagnosed with DON in the left eye and central serous chorioretinopathy (CSC) in the right eye. Considering that glucocorticoids are contraindicated for CSC and that the patient refused orbital surgery, we opted for intravenous tocilizumab (6 doses of 8 mg/kg every 4 weeks), a monoclonal antibody against the interleukin-6 receptor. After tocilizumab infusion, the disease severity in the left eye improved from DON to moderate to severe GO, with magnetic resonance imaging showing a considerable reduction in inflammation in all affected muscles. Moreover, no adverse effects were observed in this patient. Similarly, two case reports and an observational study including nine patients with DON showed good clinical results after tocilizumab infusion, with no adverse effects having been observed. Our patient was primarily treated with tocilizumab just like one of the previous cases who had uncontrolled diabetes. Tocilizumab could potentially be considered one of the treatment options for DON patients, especially those in whom glucocorticoid therapy is inappropriate. Nonetheless, high-quality studies are warranted to verify the indications for this treatment. LEARNING POINTS: Not all patients with DON are suitable candidates for or can tolerate intravenous methylprednisolone infusion. Tocilizumab, a monoclonal antibody against the interleukin-6 receptor, has been recommended as the second-line option for patients with active moderate to severe steroid-resistant GO. Tocilizumab could potentially be considered one of the treatment options for DON patients, especially those in whom glucocorticoid therapy is inappropriate.