CASE REPORT: The child was diagnosed with choroid plexus carcinoma (CPC) at age 8 months. The tumor was positive for TP53 genomic alteration. Germline genetic test sent at age 10 months returned positive for heterozygous TP53 pathogenic mutation. She underwent surgical resection, chemotherapy, and repeat resection at 13 months. At 31 months, she had three small nodules concerning for recurrent disease surrounding the large surgical cavity. Repeat surgical resection was declined by her family. She therefore underwent stereotactic radiosurgery (SRS) to each lesion to a dose of 24 Gy in 1 fraction. Anesthesia was required at the time of planning CT simulation and at the time of treatment. At one year since the procedure, she has demonstrated no further recurrent disease and no evidence of secondary malignancy at this time. DISCUSSION: Retrospective studies approximate rates of p53 mutations in children with CPC at 40%. Radiation is avoided—unless absolutely necessary—in children less than 3 years old because of the risks of neurocognitive delay, endocrine and growth toxicities, and the risk of secondary malignancies. Patients with Li Fraumeni syndrome have further dramatic increase in rates of developing secondary malignancies after exposure to ionizing radiation. SRS provides dose escalation while minimizing ionizing radiation dose to normal surrounding tissue. CONCLUSIONS: Any child diagnosed with CPC should undergo genetic testing for p53 germline mutation because of high pre-test probability. In selected cases, SRS provides an alternative to surgery as local control for limited CPC. SRS minimizes the dose of ionizing radiation delivered to normal tissues in patients with p53 mutation, or in young patients in whom less-focused radiation techniques may cause increased negative neurocognitive effects. Because of the short treatment course, SRS also minimizes the number of days under anesthesia required for young patients, thereby minimizing risk incurred by repeated anesthetic requirements.