First-degree relatives (FDR) of patients with colorectal cancer have a higher risk of developing colorectal cancer than the general population. For this reason, screening guidelines recommend colonoscopy every 5 or 10 y, starting at the age of 40, depending on whether colorectal cancer in the index-case is diagnosed at <60 or ≥60 y, respectively. However, studies on the risk of neoplastic lesions are inconclusive. The aim of this study was to determine the risk of advanced neoplasia (three or more non-advanced adenomas, advanced adenoma, or invasive cancer) in FDR of patients with colorectal cancer compared to average-risk individuals (i.e., asymptomatic adults 50 to 69 y of age with no family history of colorectal cancer). This cross-sectional analysis includes data from 8,498 individuals undergoing their first lifetime screening colonoscopy between 2006 and 2012 at six Spanish tertiary hospitals. Of these individuals, 3,015 were defined as asymptomatic FDR of patients with colorectal cancer (“familial-risk group”) and 3,038 as asymptomatic with average-risk for colorectal cancer (“average-risk group”). The familial-risk group was stratified as one FDR, with one family member diagnosed with colorectal cancer at ≥60 y (n = 1,884) or at <60 y (n = 831), and as two FDR, with two family members diagnosed with colorectal cancer at any age (n = 300). Multiple logistic regression analysis was used for between-group comparisons after adjusting for potential confounders (age, gender, and center). Compared with the average-risk group, advanced neoplasia was significantly more prevalent in individuals having two FDR with colorectal cancer (odds ratio [OR] 1.90; 95% confidence interval [CI] 1.36–2.66, p < 0.001), but not in those having one FDR with colorectal cancer diagnosed at ≥60 y (OR 1.03; 95% CI 0.83–1.27, p = 0.77) and <60 y (OR 1.19; 95% CI 0.90–1.58, p = 0.20). After the age of 50 y, men developed advanced neoplasia over two times more frequently than women and advanced neoplasia appeared at least ten y earlier. Fewer colonoscopies by 2-fold were required to detect one advanced neoplasia in men than in women. Major limitations of this study were first that although average-risk individuals were consecutively included in a randomized control trial, this was not the case for all individuals in the familial-risk cohort; and second, the difference in age between the average-risk and familial-risk cohorts. Individuals having two FDR with colorectal cancer showed an increased risk of advanced neoplasia compared to those with average-risk for colorectal cancer. Men had over 2-fold higher risk of advanced neoplasia than women, independent of family history. These data suggest that screening colonoscopy guidelines should be revised in the familial-risk population. Colorectal cancer kills about 700,000 people worldwide every year. This common cancer develops when one of the epithelial cells that lines the colon (the final part of the digestive system; also known as the large intestine or large bowel) or the rectum (the lower end of the colon) acquires genetic changes that allow it to divide uncontrollably to form a tumor (lump), to invade surrounding tissues, and to move around the body (metastasize). Symptoms of colorectal cancer include weight loss, blood in the stool, and changed bowel habits. Treatments for the condition include surgery, chemotherapy, and radiation. As with all types of cancer, these treatments are more likely to be successful if started when the tumor is small and before it has invaded surrounding tissues. Consequently, many countries run screening programs to detect the earliest signs of colorectal cancer in apparently healthy people. One of the methods used to screen for colorectal cancer is colonoscopy—visual examination of the colonic lining using a tube-like instrument that is inserted through the rectum into the colon. Colonoscopy also detects adenomas (polyps), benign tumors that lack the ability to invade or metastasize. It is important to find and remove adenomas because these growths can develop into colorectal cancers. In the United States, colonoscopy is recommended every 10 y for everyone in the general population over 50 y old. However, the first-degree relatives (parents, siblings, or child) of patients with colorectal cancer have a higher risk of developing colorectal cancer than people in the general population. So, current screening guidelines in the US and elsewhere recommend colonoscopy every 5 or 10 y from age 40 for people who have a first-degree relative diagnosed with colorectal cancer, depending on whether the relative’s colorectal cancer was diagnosed before or after the age of 60, respectively. Notably, no large-scale studies have compared the risk of adenomas in symptom-free people with first-degree relatives with colorectal cancer with the risk in symptom-free average-risk people. This large multicenter cross-sectional study (a study that looks at a single time point) aims to remedy this deficit in the evidence base. The researchers enrolled 3,015 first-degree relatives of patients with colorectal cancer and 3,038 average-risk individuals, all of whom were undergoing their first screening colonoscopy in Spain. They divided the familial-risk cohort into two groups. The 1-FDR group comprised people with one first-degree relative diagnosed with colorectal cancer at ≥60 y or at <60 y. The 2-FDR group comprised individuals with two first-degree relatives diagnosed with colorectal cancer at any age. They then compared the risk of advanced neoplasia (three or more non-advanced adenomas, advanced adenoma, or invasive cancer) in the different groups after adjusting for age and sex (confounding factors that affect an individual’s risk of colorectal cancer). Advanced neoplasia was more prevalent (present in a higher proportion of the population) in the 2-FDR group than in average-risk individuals; individuals in the 2-FDR group were nearly twice as likely to have advanced neoplasia as average-risk individuals. Compared to average-risk individuals, individuals with 1-FDR diagnosed at ≥60 y and at <60 y had no increased risk of advanced neoplasias. Notably, after age 50, men developed advanced neoplasias over 2-fold more frequently than women, and advanced neoplasias appeared at least 10 y earlier in men than in women. These findings suggest that the risk of advanced neoplasia is markedly increased in individuals having two first-degree relatives with colorectal cancer compared to average-risk individuals but not in individuals having only one affected first-degree relative. They also suggest that men have a 2-fold higher risk of advanced neoplasia than women, independent of family history. Several aspects of the study design may affect the accuracy of these findings. For example, the average-risk study participants were older than the participants with first-degree relatives with colorectal cancer, and different approaches were used to enroll the two sets of participants, which may have introduced some bias. Nevertheless, these findings suggest that the guidelines for screening for colorectal cancer should be revised. Specifically, they suggest that men with two first-degree relatives with colorectal cancer may benefit from early screening colonoscopy but that the screening strategy recommended for the average-risk population may be appropriate for individuals with only one first-degree relative with colorectal cancer. This list of resources contains links that can be accessed when viewing the PDF on a device or via the online version of the article at http://dx.doi.org/10.1371/journal.pmed.1002008.