Early-Onset Vogt-Koyanagi-Harada-Like Syndrome After Combined Ipilimumab and Nivolumab for Metastatic Melanoma: A Case Report.

Vogt-Koyanagi-Harada (VKH)-like syndrome is a rare immune-related adverse event associated with immune checkpoint inhibitors (ICIs), resulting from an autoimmune response directed against melanocyte-associated antigens. We report the case of a 56-year-old woman with stage IV metastatic melanoma who developed fever, diffuse pruritic lichenoid dermatitis, and acute bilateral visual loss one week after one cycle of combined ipilimumab and nivolumab. Ophthalmological evaluation demonstrated bilateral serous retinal detachments consistent with VKH-like syndrome, while cerebrospinal fluid analysis confirmed aseptic lymphocytic meningitis. Skin biopsy revealed lichenoid interface dermatitis associated with a mild reduction in melanocyte density demonstrated by Melan-A and SOX10 immunostaining. Immunotherapy was discontinued, and treatment with high-dose systemic corticosteroids followed by methotrexate resulted in rapid clinical improvement. More than two and a half years after permanent discontinuation of ICI therapy, the patient remains in sustained complete radiological remission without resumption of immunotherapy. This case illustrates an uncommon presentation of early-onset VKH-like syndrome following dual immune checkpoint blockade, combining ophthalmologic, neurological, and histologically documented cutaneous involvement. It also highlights the importance of early multidisciplinary management to preserve visual function while optimizing oncological care. Although VKH-like syndrome cannot currently be considered a validated predictive biomarker, accumulating evidence suggests that melanocyte-directed autoimmunity may reflect a particularly effective antitumor immune response in a subset of patients with metastatic melanoma.