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PurposeTo describe a case of a patient who presented with hypertensive retinopathy and was found to have neuroblastoma.ObservationsNeuroblastoma has three main ocular presentations. As a primary disease, it can present with a paraneoplastic syndrome in the form of opsoclonus, or it can present as a Horner's syndrome from its effect on the cervical sympathetic ganglia. Metastatic disease can present as a triad of periorbital edema, ecchymosis and proptosis from orbital bone involvement. Hypertension is a rare systemic presentation of neuroblastoma. We report the case of a two-year-old girl whose initial presentation of neuroblastoma was hypertensive retinopathy.Conclusions and importanceTo our knowledge, this is the first reported case of neuroblastoma presenting as hypertensive retinopathy. Our case highlights the importance of a multidisciplinary approach and thorough systemic work up of ocular findings in order to arrive at an appropriate diagnosis.