Introduction: Malaria is a public health concern and still endemic in Malaysia particularly in Sabah. Malaria is due to plasmodium parasite which can be further divided into human malaria and zoonotic malaria. Plasmodium knowlesi, a zoonotic malaria species, is increasing in trend and alarming despite of ongoing prevention and control measures especially in Sabah. Methods: This is a case-control study involving a total of 169 respondents which comprises of 43 cases and 126 controls from Mukim Paginatan and Mukim Nalapak, Ranau. A simple random sampling were used to select subjects. Results: This study found male, forest related and doing outdoor activity has higher risk of contracting malaria. Village house is another significant risk factor. The specific risk factors with the strongest association with malaria is the presence of monkey near housing or farm area (OR5.667; 95% CI: 2.429, 13.220), live near forest (OR 5.330; 95% CI: 1.209, 23.495), presence of pond or pool or river near house (OR 4.222; 95% CI: 1.552, 11.490), doing outdoor activity after 5 PM (OR 3.434; 95% CI: 1.522, 7.748) and ever overnight in the hut (OR 2.724; 95% CI: 1.334, 5.561). Conclusion: This study shows several risk factors that associated with malaria knowlesi. Based on the identified risk factors, preventive and control measure can be improved and optimized.