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Background and aim Congenital leukemia is an extremely rare condition, with an estimated incidence of 1–5 per 1,000,000 live births, characterized by an aggressive clinical course and poor prognosis. Case presentation We report a case of congenital leukemia in a 1-month-old male infant with an unremarkable family and antenatal history, admitted to the intensive care unit in a comatose state (Glasgow Coma Scale score of 3) with suspected intracranial hemorrhage. Laboratory evaluation revealed a WBC count of 1,000 × 10 9 /L with 99% blast cells. The clinical course was complicated by severe hemorrhagic manifestations, including multiple intracerebral hemorrhages and cerebral edema. Despite multidisciplinary supportive management involving pediatricians, neonatologists, neurologists, and neurosurgeons, cytoreductive procedures such as leukapheresis or exchange transfusion could not be undertaken because of hemodynamic instability, active intracranial bleeding, and resource limitations, and the patient died within 24 h of admission. The diagnosis of acute myeloid leukemia with a KMT2A (MLL) rearrangement was confirmed postmortem. Conclusions Our case demonstrates the aggressive nature of congenital leukemia and its potential to present with life-threatening hemorrhagic complications. Early recognition and rapid diagnostic evaluation are essential; however, prognosis remains poor in fulminant cases. Multidisciplinary management is critical for timely decision-making and supportive care. This report emphasizes the importance of considering congenital leukemia in neonates presenting with unexplained coma and hyperleukocytosis, even in the absence of typical clinical signs.
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Tugelbayeva et al. (2026) studied this question.
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