Thalassaemia is one of the most common autosomal recessive blood disorders in the world and its carrier status is prevalent in nearly 15% of Malaysian population. The global and economic burden for lifelong care of those affected increases every year. Currently, there is no policy on thalassaemia-carrier screening for couples prior to marriage besides HIV/AIDS screening in Malaysia. Other countries such as Iran, Iraq, Turkey, Bahrain and Saudi Arabia have established a policy for thalassaemia prevention by conducting premarital thalassaemia screening. Zero thalassaemia cases in new-born child in Cyprus have proven that thalassaemia can be prevented. This study aimed to investigate the willingness of premarital Malays on premarital thalassaemia screening. A set of questionnaire was distributed to 57 persons at premarital course sites and wedding fairs and expositions held in Kuala Lumpur and Selangor. Components in the questionnaire included: 1) demographic 2) knowledge about thalassaemia, signs and screening method 3) attitudes towards thalassaemia premarital screening 4) practices of premarital thalassaemia screening. Analysis for the questionnaire was performed using IBM SPSS Statistics 23.0. A majority of the participants (75.44%) have never heard of thalassaemia as a medical condition. However, at least half of the participants (56.14%) agreed that premarital screening is needed to know their status of thalassaemia. In terms of facilities provided, widely covered campaigns about facilities of the screening test is also one of the factors that could encourage people to do premarital thalassaemia screening (35.09%). Even though premarital thalassaemia screening is not an obligation, 50.88% of participants agreed that it is important. The main reason people refused to do premarital thalassaemia screening was because they were afraid to know the test result and how it may affect their lives. Although preliminary, we have demonstrated that the level of knowledge concerning thalassemia among Malay Malaysians is still low. This may contribute to the low awareness about the effects of the disease and the importance of premarital thalassemia screening. However, most participants agreed that it is beneficial to perform the premarital thalassemia screening to identify individuals at risk of this disease. Nevertheless, many factors need to be taken into consideration if premarital thalassaemia screening is to be implemented in Malaysia as a national policy.