Objectives: To remind that despite its low incidence, leukemic relapse in the testicles worsens the prognosis of this haematopoietic neoplasia.
Methods/results: We report the case of a 20-year-old male suffering from acute lymphoblastic leukemia in complete remission after induction and maintenance treatment, who suffered a leukemic relapse in both testicles. Diagnosis was made by ultrasound and confirmed after bilateral testicular biopsies. The patient died due to fungal infection after new treatment with systemic chemotherapy and bone marrow transplantation.
Conclusions: It is necessary to suspect the existence of leukemic cells within the testicles in patients with this disease, mainly acute lymphoblastic leukemia, who present with acute testicular symptoms. Testicular biopsy confirms the diagnosis. Treatment is systemic, with testicular radiation to eradicate the leukemic reservoir. Despite the achievement of local disease control, this situation worsens the prognosis.
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