Klinefelter’s syndrome, 47, XXY, is the most common chromosomal aberration among men and the incidence of the syndrome is 2.0 per 1,000 live-born males. Individuals with XXY may have hypogonadism, fertility problems, tall stature, gynecomastia, language based learning disabilities, and disorders of executive function. It is found that language disorder is seen in 65% as the most prevalent disorder followed by attention deficit disorders in 63% and autism spectrum disorder 27% of all cases suffering this syndrome. Behavioral impairment is most evident among cases classified as autism spectrum disorder and psychotic disorder with a rate of 12%. Although there are more reports about Klinefelter’s syndrome associated with psychiatric disorders in literature, there are limited reports regarding personality traits. We present here a case report of Klinefelter’s syndrome, who has been suffering antisocial personality disorder. A 17-year-old male was admitted to the emergency ward because of aggressive behavior, substance use and self mutilation. He started using alcohol since he had been 8 years old. He started to use cannabis when he was 11 years old. He has had impulsive behaviors since childhood. He reportedly had multiple self-inşicted injuries. His psychiatric history was significant for the diagnosis of attention deficit and hyperactivity disorder and conduct disorder. Physical examination indicated that he was a tall (200 cm) and overweight (110 kg) boy with long arms and legs. We was suspected to have Klinefelter’s syndrome due to his physical appearance. Cytogenetic studies showed a 47, XXY karyotype. All other investigations gave results within normal limits. His intellectual function scores on the Wechsler scale were 77 for performance, 86 for verbal skills and 83 in full scale. Klinefelter’s syndrome is associated with multiple psychiatric comorbidities. Recent studies on the genetics of alcoholism have suggested an association between antisocial behaviors and the MAO-A gene. It is found that the rate of MAO-A promoter polymorphism was 3% among type 2 alcoholics. They suggest that MAO-A is X linked; the heterozygotes are probable cases of Klinefelter’s syndrome (47, XXY) suggesting that X-chromosome aneuploidy might increase the risk for developing type 2 alcoholism. Previous studies consistently demonstrate that for men, and probably for women, a history of conduct disorder in childhood and adulthood might be an indicator of predisposition to the development of an alcohol misuse.