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Objective: To arouse the suspicious for early diagnosis and hence, proper management of megameatus with an intact prepuce (MIP), as there is no external clue for detection of such cases, which usually come to light for the first time in a boy who is about to retract his prepuce or during neonatal circumcision., Materials and Methods: Examination of neonates and infants coming to circumcision clinic to detect congenital genitourinary anomalies. Evaluation of 12,518 neonates and infants coming for ritual circumcision from 2006-2017, who were examined thoroughly to detect any incidental congenital genitourinary anomalies. Fifteen of them were diagnosed to have a MIP anomaly. They were investigated to perceive any associated median raphe (MR) anomalies. Sensitivity, positive predictive value, specificity, and negative predictive value of MR anomalies in cases of MIP were estimated and compared with other children who had a normally positioned meatus., Results: Overall incidence of MIP in this group of babies was 0.12%. Twelve of 15 cases (80%) with MIP had 19 forms of MR anomalies; mainly raphe deviation in 6 cases, hyperpigmented raphe in 6, prominent raphe in 4, and bifurcation in 3 cases. Three cases had a redundant long prepuce, and 1 had paraphimosis after preputial retraction, otherwise no other genitourinary anomalies could be detected in those cases., Conclusion: MR anomalies, mainly deviation and hyperpigmented prominent raphe, are significant indictors for the presence of an invisible MIP anomaly. Abnormally redundant long prepuce may be seen in such cases, but this is not common., (Copyright © 2018. Published by Elsevier Inc.)