Benign meningiomas are characterized by a normal karyotype or loss of all or part of chromosome 22. Histologically higher grade tumors are typically characterized by a pattern of increasing chromosome loss and instability. This characteristic pattern of unbalanced chromosome aberrations is punctuated in the literature by several intriguing reports of a reciprocal t(1;19)(q21;q13.3) as the sole cytogenetic aberration. We report a third case showing the t(1;19)(q21;q13.3) with additional unstable secondary aberrations of a dic(18;22)(p11;p11) and telomeric fusions.