Background: Gender-specific risk factors have been suggested to promote a fourfold higher incidence of pilonidal sinus disease (PSD) in male as compared to female patients. However, in recent decades there has been an apparent shift towards an increasing prevalence of PSD in women, as body weight and other risk factors influence the disease. We aimed at determining whether PSD prevalence actually changed in men and women over time. Methods: Following PRISMA guidelines (PROSPERO ID: 42016051588), databases were systematically searched. Papers reporting on PSD published between 1833 and 2018 in English, French, German, Italian and Spanish containing precise numbers of male and female participants were selected for analysis. Gender-specific prevalence of PSD over several decades was the main outcome measure. Results: We screened 679 studies reporting on 104 055 patients and found that the male/female ratio in patients with PSD has remained constant over time, with women being affected in about 20% of all PSD cases (I 2 = 96.18%; meta-regression p < 0.001). Conclusion: While the prevalence of PSD has risen over the past decades, the ratio between affected males and affected females has remained constant, with women invariably representing about 20% of patients despite wide ranging socioeconomic and behavioural changes.