Abstract It is common belief that in families with hereditary prostate cancer (HPC), unaffected men should be screened periodically with PSA, but little is known about the effects of such screening. We studied test and tumor characteristics in unaffected 50–75‐year‐old screenees from HPC families. In the Netherlands, 153 verified HPC families are registered; 132 unaffected men in these families were not under surveillance for prostate cancer and gave informed consent for PSA testing by their GP and referral to a urologist in the case of a PSA level ≥ 3.0 ng/ml. Results were compared to published data from the Rotterdam and Göteborg sections of the European Randomized Study of Screening for Prostate Cancer (ERSPC). A PSA ≥ 3.0 ng/ml was found in 20 men: referral rate, 15.1% (ERSPC Rotterdam: 20.1%; ERSPC Göteborg: 12.0%). Only 3 cases of prostate cancer were diagnosed in these men: detection rate in the first screening round 2.3% (ERSPC Rotterdam: 5.3%; ERSPC Göteborg: 2.3%). Frequent opportunistic PSA testing made it impossible to estimate the detection rates in subsequent screening rounds. In the first and subsequent PSA screening rounds, 11 cases of cancer were detected. All but 1 had favorable tumor characteristics (cT1c/pT2; Gleason < 7). These results raise the question as to whether men from all HPC families should be considered at high‐risk. We suggest that the same PSA testing guidelines should apply to HPC families and the general population. A more aggressive screening policy in HPC families does not seem to be justified. © 2007 Wiley‐Liss, Inc.