Background : Acute leukemia is typically identified through clinical signs of cytopenia and/or a tumor syndrome, with paraneoplastic syndromes being much rarer. Procedure : We describe a case of galactorrhea and amenorrhea as first symptoms in a child with acute myeloid leukemia (AML). We reviewed the literature and explored potential mechanisms of prolactin (PRL) production. Results : We present a case of a 10-year-old girl who initially presented with inflammatory joint pain, night sweats, weight loss, amenorrhea, along with breast swelling and galactorrhea. At diagnosis, her PRL level was elevated (260.5 μg/L-reference:< 25 μg/L), and she exhibited biological hypogonadism. Blood counts revealed anemia, thrombocytopenia, and 11% circulating blasts. Bone marrow aspiration confirmed AML, classified as FAB M5, with a KAT6A::CREBBP fusion transcript. Cerebrospinal fluid analysis was negative for blasts, and brain magnetic resonance imaging showed no leukemic infiltration of the pituitary gland or concomitant pituitary tumor. Notably, PRL level normalized following chemotherapy. In the absence of central nervous system involvement, ectopic PRL secretion by leukemic blasts is the most plausible explanation for the elevated PRL levels in this case. Conclusion: Acute leukemia is a rare cause of hyperprolactinemia, which can result from various mechanisms including ectopic synthesis by blasts, hypothalamic-pituitary infiltration, stress, or iatrogenic factors. It is crucial to perform hypothalamic-pituitary magnetic resonance imaging and lumbar punction are essential to rule out central nervous system involvement and suprasellar region infiltration. PRL levels might serve as potential markers for monitoring treatment response and detecting relapse in cases presenting with initial hyperprolactinemia.