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Introduction: Dorsal penile frenulum is a very rare abnormality in literature. Case Report: This abnormality was present with normal external urethral meatus in a young adult male. There was no associated penile torsion. Conclusion: This is the second case report in medical literature. A brief description of preputial anatomy and frenar band band is described. Various hypotheses have been discussed for development of dorsal penile frenulum.

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