Background: The purpose of this study was to determine the prevalence of allele and genotype variants of follicle-stimulating hormone receptor (FSHR) gene polymorphic region at position Asn680Ser in the Albanian male population and associate them with the clinical parameters of infertility. A total of 123 infertile men (35.04±5.85 years) stratified according to the level of spermatogenetic impairment (oligoasthenozoospermia, asthenozoospermia, and azoospermia), and 112 fertile men (36.44 ±7.05years) with normal semen parameters were enrolled in this study. ----- Methods: Genotyping of the FSHR gene at position 680 was performed by TaqMan genotyping assay. All participants underwent semen analysis; serum reproductive hormones (FSH, luteinizing hormone, prolactin, and testosterone) were also measured. ----- Results: The FSHR Asn680Ser genotype frequencies were 42% Asn/Ser, 33.9% Ser/Ser, and 24.1% Asn/Asn for the control group, and 55.3% Asn/Ser, 25.2 % Ser/Ser and 19.5% Asn/Asn for the overall infertile men group (P = 0.08,χ2 test). No statistically significant correlation between serum hormonal levels and semen characteristics or fertility status and FSHR Asn680Ser gene variants was found either in control subjects or the infertile men group. However, adjusted logistic regression analysis (age, body mass index, smoking, and alcohol as covariates) revealed increased odds ratio for male infertility among heterozygous Asn/Ser genotype carriers associated with lower values of semen parameters (normal morphology, concentration, total sperm count, and motility). ----- Conclusion: In conclusion, our case-control study further confirmed previous reports of no significant association between the FSHR Asn680Ser polymorphisms and male infertility. Nevertheless, the data presented here indicate that Asn/Ser genotype may increase the risk for male infertility in the Albanian population of the Dukagjini Region.