The peptide hormones of the adenohypophysis are produced by proteolytic processing of their prohormone precursors. Cathepsin L is known to function as a major proteolytic enzyme involved in the production of the peptide hormones. The structure of the propeptide region of cathepsin L is identical to cytotoxic T-lymphocyte antigen-2 alpha (CTLA-2α) which is also shown to exhibit selective inhibitory activities against cathepsin L. However, the specific cell types synthesizing CTLA-2α in mouse adenohypophysis and its functional implications as relevant in vivo have not been demonstrated. In this study, CTLA-2α expression in the adenohypophysis was evaluated by immunohistochemistry. In both male and female mice, strong immunoreactivity was specifically detected in folliculostellate (FS) cells surrounding endocrine cells which were delineated by CTLA-2α. These findings suggest that the CTLA-2α may be involved in the proteolytic processing and secretion of the hormones in the adenohypophysis through regulation of cathepsin L.