Testicular dysmorphism (TD) associated with infantile abdominoscrotal hydrocele (ASH) may be due to the pressure effect of hydrocele on the testis or may be a developmental abnormality. Only a few reports are referred to the etiology of testicular changes in infantile ASH. We present an additional case of infantile ASH together with a review of the literature, focusing on insights into etiology of TD and its appropriate management.
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