Background: Inadequate use of statistics in biomedical research might not only affect science but also harm human beings if applied in medical practice. Biostatistics is fundamental to improve understanding and appraising of evidence-based medicine (EBM); yet, it is still not well understood and appreciated by medical students. Therefore, early exposure of medical students and physicians-in-training to research tools including Biostatistics is of utmost importance. Objective: The aim of this study is to determine the perception of Biostatistics by medical students at a private medical school in Beirut, Lebanon, and to identify its best implementation time in the medical curriculum. Methods: This is a cross-sectional study based on a self-administered questionnaire distributed among medical students in their pre-clerkship years (first three years of a 6-year program) who undertook Biostatistics. The assessment of perception was based on the 5-point Likert scale anchored by Strongly disagree = 1 and Strongly agree = 5 including 36 questions distributed into four domains to assess the course value, difficulty, behavioral, and expectations. Results: 186 of 269 students responded to the questionnaire, yielding a response rate of 69.14%. Around 60% of students declared that the knowledge gained from biostatistics courses is useful to their future career, and almost 70% understood the main concepts of biostatistics. 57.7% of students perceived that lack of practicing exercises might contribute to making the course more difficult. The mean score of domains was higher in females but did not significantly differ within the three academic years. Only 35.1% of the students positively perceived the importance of biostatistics modules, mostly third-year students. Conclusion: Although the majority of medical students perceived biostatistics modules negatively, they were aware of the relevance of biostatistics to their medical career and real-life health issues.