Introduction: Hemorrhagic cystitis in the newborn is a very rare occurrence and is primarily caused by a secondary disease. However, there is scarcity of data depicting its actual incidence. Although uncommon, neonatal gross hematuria can be a manifestation of a potentially serious pathology . As such, gross hematuria should be evaluated promptly . Case presentation: We report a rare case of term male neonate who presented with gross hematuria seen immediately after birth. Hematuria was confirmed with urine microscopy . Both of his blood and urine culture were negative for microbial growth. His coagulation profile was unremarkable. Anemia and thrombocytopenia, which were probably due to acute blood loss, were observed on full blood count. His renal tract ultrasound showed thickened bladder wall with inflammatory or hemorrhagic debris but no other structural abnormalities. He received intravenous vitamin K, antibiotics, and blood transfusion, and was managed conservatively . His hematuria resolved on the eighth day of life without further episodes until 2-month of life, with no clear cause of the presentation. Conclusion: In this paper, we discuss a rare case of neonatal hemorrhagic cystitis and its clinical approach. Moreover, this reinforces the importance of distinguishing hemorrhagic cystitis from other more serious conditions that present with gross hematuria. This underscores the necessity of prompt evaluation and appropriate intervention to achieve good clinical outcome.