• Cédric Baltus, Sébastien Toffoli, Frédéric London, Paul Delrée, Claude Gilliard, and Thierry Gustin.
• Department of Neurosurgery, CHU UcL Namur, Yvoir, Belgium. Electronic address: cedricbaltus.pro@gmail.com.
• World Neurosurg. 2019 Oct 1; 130: 380-385.

BackgroundChromothripsis is characterized by a multitude of chromosomal rearrangements during a unique cataclysmic event in a cell life. Disintegration of one or several chromosomes is followed by a chaotic rearrangement of generated fragments. It might play a role in oncogenesis and tumor progression. It is observed in 2%-3% of cancers and is rarely reported in benign tumors. We report a case of massive chromothripsis in a fast growing chordoid meningioma.Case DescriptionA 55-year-old woman was admitted for a meningeal mass developing in the right parietal parasagittal area. She underwent subtotal resection of the tumor. Histologic analysis revealed a chordoid meningioma (World Health Organization grade II). Six months later, magnetic resonance imaging showed a large bilateral tumor recurrence. After a second surgery, the patient received radiotherapy. Thereafter, the clinical course was uneventful. Comparative genomic hybridization showed only a monosomy X in the primary tumor. In the recurrent meningioma, this anomaly was associated with a massive chromothripsis including more than 370 chromosomal abnormalities affecting chromosomes 1-22.ConclusionsChromothripsis is rarely described in benign tumors and especially in meningiomas. In the presented case, the high number of chromosomal rearrangements and the onset of this phenomenon at a later stage of tumor progression are very unusual. The role of surgical stress on the emergence of chromothripsis and its relation with tumor aggressiveness are discussed.Copyright © 2019 Elsevier Inc. All rights reserved.

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