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Involvement of the prostate gland, as an early extra-nodal manifestation of a hematologic disease, or as a secondary infiltration is rare. Even rarer is the acute urinary retention due to infiltration by lymphocytes and simultaneously enlarged prostate. We present a case of a 61 years old male patient with a history of chronic lymphocytic leukemia, who was under oncological follow-up with no active treatment and had typical lower urinary tract symptoms due to benign prostatic hyperplasia and was receiving 5-alpha reductase inhibitor. After an acute urinary retention episode which was managed with a suprapubic catheter due to urethral catheter insertion failure, the patient was submitted to a transurethral prostatectomy. Histological examination revealed lymphocytic infiltration of the prostatic parenchyma by mostly small B cells. B-lymphocytic infiltration of the prostate gland, causes symptoms similar to benign prostatic hyperplasia. Acute urinary retention due to B-lymphocytic infiltration of the prostate is rare and the diagnosis is always histological and an oncological re-evaluation is necessary. The prognosis of these patients is related to the generalized disease rather than to the prostatic involvement.
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