Sex cord stromal tumors with annular tubules (SCTAT) are rare neoplasms typically found in the ovary and are often associated with Puetz-Jeghers syndrome. However, SCTAT arising in testicular tumors are exceedingly rare, only with five cases documented in literature. These tumors display unique histological characteristics. We report an unusual case seen in a 25 year old male who presented with bilateral undescended testes. Right sided orchidectomy with left sided orchidopexy was done. Histopathological examination revealed SCTAT with diffuse cytoplasmic positivity for inhibin and calretinin on immunohistochemistry. The patient exhibited no features of Puetz-Jegher’s Syndome. He is currently under surveillance. This case underscores the significance of SCTAT in testicular tumors, particularly in patients with a history of undescended testes.
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