Meningiomas were among the first solid tumors recognized as having cytogenetic alterations. The most consistent changes reported in grade I meningiomas were monosomy 22 or partial 22q deletion. The vast majority of meningiomas are histologically benign, but the prognosis is determined by risk of recurrence after surgical treatment. Despite important advances in the identification of prognostic factors in the past decade, the exact nature of tumor recurrence remains largely unknown. In the present study, a recurrent transitional meningioma deriving from the anterior portion of the falx cerebri was characterized by combining conventional cytogenetics and array comparative genomic hybridization (CGH). Cytogenetic analysis at diagnosis revealed the following complex numerical and structural aberrations: 42,XY,der(1)t(1;?)(p12;?),-6,der(12;15)(q10;q10),-18, -22. Additional clonal evolutions were further identified with disease relapse. Array CGH corroborated the cytogenetic findings. The presence of a complex cytogenetic profile and progression-associated chromosomal abnormalities in a benign meningioma suggests the existence of underlying molecular events.