Introduction Neuroblastoma is the most common extracranial solid tumor in pediatrics but is considerably uncommon in adults, with approximately 1 case per 10 million diagnosed per year and is associated with poor prognosis. There are no standard treatment protocols for adult-onset neuroblastomas and there are only a few published case reports on neuroblastoma in adults. Case report We report our treatment experience in a 41-year-old female diagnosed with high-risk, poorly differentiated neuroblastoma. Management and outcome Our patient received two cycles of dinutuximab adapted from the Children’s Oncology Group ANBL1221 protocol. The patient experienced pain, neuropathy, pruritus, and infusion reactions which were managed with supportive care. Due to the lack of tumor regression, dinutuximab was omitted from future treatments. Currently, the patient is asymptomatic from her disease and remains off of all therapy and pain medication. Discussion While dinutuximab has produced promising outcomes in pediatric patients, it is not without potentially severe adverse effects. Serious reactions of capillary leak syndrome, infusion reactions, pain, and neuropathy have been reported. Clinicians must be cognizant of the treatment-related toxicities associated with dinutuximab therapy, ranging from pain, neuropathy, pruritus, and infusion reactions as explored in this patient case.