We present a case of acute myeloid leukemia (AML-M2) with trisomy 5 (+5) as the sole cytogenetic abnormality in a woman previously diagnosed with schizophrenia. To date, only two cases of AML (other than M2) with +5 as the only change have been reported. Moreover, an association between schizophrenia and partial trisomy of chromosome 5p has been described recently. To our knowledge, this is the first report of AML (subtype-M2) with +5. Noteworthy is the association with schizophrenia.