Background: A varicocele is abnormal dilatation and expansion of the scrotal venous pampiniform plexus which drains blood from each testicle. While typically painless, varicoceles are clinically relevant since they are the most often recognized cause of infertility. The specific process by which varicocele causes infertility remains unclear. It is widely known that the pathophysiology of varicocele caused infertility is complicated and multifaceted. The severe damage produced by varicocele is connected with reduced function of sertoli cells. AMH has been considered as a direct marker of Sertoli cell activity and an indirect marker of spermatogenesis. So with prolonged varicocele , assessment of serum AMH be one of diagnostic studies in varicocele patients. Also, following varicocelectomy examination of percentage of success and reversal of normal tistecular function is an essential factor for raising of pregnancy rate. The purpose of this research was to analyze the serum level of antimullerian hormone in varicocele patients before and after varicocelectomy and evaluation of its connection to semen parameters and varicocele grade. Methods: This case control research was done on thirty varicocele patients and twenty age and sex matched healthy volunteers as controls. They were recruited from the outpatient clinic of Dermatology, Venereology and Andrology Department of Benha University hospitals from Dec 2019 to sept 2020. Results: There was non significant difference was noticed between patients and control groups as respects age. Varicocele grade showed substantial improvement post-operative. Before varicocelectomy, sperm count and motility was considerably lower in patients than control. In contrast, aberrant forms was much greater in patients than control. The mean sperm count and motility considerably higher after than before varicocelectomy. In contrast, aberrant forms dramatically diminished after than before varicocelectomy. AMH level in patients before varicocelectomy was substantially lower than in controls whereas after varicocelectomy AMH level rose. Before varicocelectomy, AMH level exhibited a substantial negative connection with varicocele grade and aberrant morphologies. After varicocelectomy, AMH level revealed a substantial positive connection with sperm count. AMH was a significant predictior of varicocele after adjusting for the influence of patients׳ age and smoking. Conclusion: The research indicated that AMH decrease in varicocele patients and rise after surgical repair compared to controls which linked with Sertoli cells alterations histopathologically and this the first study evaluated AMH after varicocelectomy. Thus, a finding that reinforces the importance of AMH as a trustworthy measure of Sertoli cell activity.