Introduction and Importance: Even though urolithiasis in general is not uncommon, urethral stones have an incidence of less than 0.3% and are 20 times less common in children. Though cases of urethral stones have been reported in children from endemic areas, they are even rarer in countries like Uganda that are not endemic for urolithiasis. Case Presentation: The authors present a 7-year-old male who presented with acute urine retention. Though the diagnosis of retention was made in a lower-level health facility, the cause of the retention was not determined till the patient arrived at a general hospital. Diagnosis of an obstructing stone in the penile urethra was made clinically. Meatotomy and stone extraction were done, and a urethral catheter was passed. Clinical Discussion and Conclusion: When attending to children with acute urine retention, urolithiasis should be kept among the differential diagnoses, even in areas that are not endemic for urinary tract stones. A thorough clinical evaluation may be all that is needed to make a diagnosis.
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