The 1st pediatric case of sertoliform cystadenoma with unique features is described herein. The patient is a 6-year-old boy who presented with gynecomastia and a left testicular cystic mass. Histopathologically the tumor was found to originate from the rete channels, filling and distending them with areas of mural Sertoli cell proliferations reminiscent of large cell Sertoli cell tumor (noncalcifying form) and showing widespread intratubular Sertoli cell proliferation islands in the vicinity. Histopathologic and immunohistochemical features are described in light of the relevant literature.
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