Cytokines are paracrine/autocrine growth factors known to affect testicular cell functions. The cellular origin and expression levels of interleukin-1 (IL-1) in human normal and pathological testicular biopsies are not yet clear. In the present study, we have investigated the levels and cellular origin of IL-1 family members [IL-1α, IL-β, and IL-1 receptor antagonist (IL-1ra)] in human testicular normal and abnormal biopsies with incomplete maturation arrest (IMA) or Sertoli only syndrome (SOS), using real-time polymerase chain reaction and immunohistochemical staining analysis. Our results show that the levels of IL-1α were higher in Leydig/interstitial cells of biopsies with IMA and SOS compared with normal. The levels of IL-1α in Sertoli cells of normal biopsies were higher than IMA and SOS. The mRNA levels of IL-1α were similar in all biopsies. IL-1β levels were higher in Leydig/interstitial cells of normal biopsies compared with Sertoli and germ cells. The levels of IL-1β were similar in testicular cells of all biopsies. However, the mRNA levels of IL-1β were significantly lower in SOS and IMA biopsies compared with normal. IL-1ra was expressed only in Leydig/interstitial cells, and their expression in normal biopsies was higher than in biopsies with IMA and SOS. The mRNA levels of IL-1ra were similar in all biopsies. Thus, it is possible to suggest the involvement of IL-1 system in the regulation of spermatogenesis and male infertility.