OBJECTIVE AND IMPORTANCE Ectopic recurrence of a craniopharyngioma is a rare postoperative complication. We present a case of a craniopharyngioma that ectopically recurred along the tract of a previous surgical route. CLINICAL PRESENTATION A 73-year-old female patient presented 8 years earlier with a suprasellar craniopharyngioma. She underwent a right frontal craniotomy, with an interhemispheric transcallosal approach, for total microsurgical resection of the tumor. No postoperative radiotherapy was administered. Four years after surgery, magnetic resonance imaging studies revealed a well-circumscribed, heterogeneously enhancing, parasagittal mass with significant vasogenic edema in the right frontal lobe. Enlargement of the lesion was noted in subsequent radiological evaluations until 8 years after surgery, when the patient experienced a significant decline in neurocognitive status and the mass was surgically resected. INTERVENTION Gross total resection of a histologically confirmed craniopharyngioma was achieved. CONCLUSION To our knowledge, only eight previous case reports described the ectopic recurrence of a craniopharyngioma. Transplantation of tumor cells along the tract of a previous surgical route in six cases and dissemination in cerebrospinal fluid in two cases are presumed to be the primary mechanisms by which these ectopic recurrences occurred. The results of our literature review led us to conclude that total surgical resection, combined with careful inspection and irrigation of the surgical field, is the optimal treatment for preventing ectopic recurrences. Furthermore, it is recommended that, after primary craniopharyngioma resection, patients undergo long-term clinical and radiological follow-up monitoring for the rare development of an ectopically recurring tumor.