For hypospadias surgeons, avoiding parental regret surrounding childhood hypospadias surgery is a clinical priority. Individual studies have cautioned otherwise. The objective of this review is to systematically evaluate the literature and establish the true prevalence of and the predictive factors for parental decision regret in hypospadias surgery; either after agreeing to or refusing operative repair for their son. We performed a systematic search of electronic databases, grey literature and citation tracking platforms with no restriction on language. Studies addressing disorders of sexual differentiation were excluded. Quality assessment was performed using the Joanna Briggs Institute’s critical appraisal tools. Three studies were included in the review (439 participants). There were no studies discussing regret in surgery for proximal hypospadias or regret after refusing hypospadias surgery. Mean prevalence of parental decision regret after consenting for distal hypospadias surgery was 80.9% (mild 49.4%, moderate/strong 31.4%); mean follow-up was 3.9 years (range 1-9 years). The significant predictors of decision regret were postoperative complications, preferences concerning circumcision, decision conflict between parents, parental educational level, patient not being the firstborn, family history of hypospadias and initial desire to avoid surgery. This review has identified both, important knowledge gaps in the area of parental decision regret after hypospadias surgery and methodological weaknesses in existing studies. Although the reported regret after consenting for distal hypospadias repair was high, risk factors associated therewith appeared to be inconsistent between studies raising the possibility of imprecision due to unknown confounders.