The present paper aimed to systematically review case studies on physique athletes to evaluate longitudinal changes in measures of body composition, neuromuscular performance, chronic hormonal levels, physiological adaptations, and psychometric outcomes during pre-contest preparation. We included studies that (1) were classified as case studies involving physique athletes during the pre-contest phase of their competitive cycle; (2) involved adults (18+ years of age) as participants; (3) were published in an English-language peer-reviewed journal; (4) had a pre-contest duration of at least 3 months; (5) reported changes across contest preparation relating to measures of body composition (fat mass, lean mass, and bone mineral density), neuromuscular performance (strength and power), chronic hormonal levels (testosterone, estrogen, cortisol, leptin, and ghrelin), physiological adaptations (maximal aerobic capacity, resting energy expenditure, heart rate, blood pressure, menstrual function, and sleep quality), and/or psychometric outcomes (mood states and food desire). Our review ultimately included 11 case studies comprising 15 ostensibly drug-free athletes (male = 8, female = 7) who competed in various physique-oriented divisions including bodybuilding, figure, and bikini. The results indicated marked alterations across the array of analyzed outcomes, sometimes with high inter-individual variability and divergent sex-specific responses. The complexities and implications of these findings are discussed herein.