ArticleJournal of neurosurgery. Case lessons2026
Lethally aggressive multiply recurrent adamantinomatous craniopharyngioma without histological malignancy: illustrative cases.
Article in Journal of neurosurgery. Case lessons, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
backgroundMortality from craniopharyngioma (CP) is rare and typically results from surgical or endocrine-metabolic complications rather than tumor growth. Rapidly lethal regrowth without malignant transformation is uncommon and poorly characterized. OBSERVATIONS: The authors report on the cases of 2 patients with multiply recurrent adamantinomatous CP (ACP)-one childhood-onset case with a late-adolescent lethal recurrence and one adult-onset case that developed rapid, treatment-refractory regrowth within months despite multiple resections, radiosurgery, and cytotoxic chemotherapy, culminating in death. Neither tumor showed histological malignant transformation. Both tumors had a Ki-67 labeling index (LI) exceeding 10% with Ki-67-positive epithelial clusters. Imaging and intraoperative findings showed firm adherence to the hypothalamus and third ventricle with a cyst-predominant pattern, suggesting that residual adherent cyst-wall epithelium may contribute to aggressive relapse. LESSONS: Multiply recurrent ACP with a high Ki-67 LI can follow a rapidly lethal course despite benign histology. Early recognition of this aggressive phenotype should prompt short-interval MRI surveillance and timely, goal-directed salvage focused on proactive cyst control, hypothalamus-sparing strategies, and carefully selected reirradiation. When standard options fail, enrollment in clinical trials of emerging biological therapies, such as IL-6 blockade, intracystic peginterferon-α2a, and EGFR-axis treatments, should be considered. https://thejns.org/doi/10.3171/CASE25840.
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