Chronic myeloid leukemia (CML) is a relatively rare malignancy of cells of the hematopoietic system. Loss of vision and hearing is a very rare presenting manifestation of CML especially at a young age. We are reporting a rare case of CML in 16 years old male presenting with loss of hearing and vision in Muhimbili National Hospital in Tanzania. This case shows how the non-communicable disease can cause morbidity in children in a developing world and the importance of early diagnosis and management of CML in prognosis. In addition the case shows how adequate investigations and documentation is important in follow up of the patient clinical conditions and treatment outcomes. The patient was put on Tabs Alluprinol 300mg once daily for one month and Tabs hydroxyl urea (HU) 3g once daily for one month. 2 weeks later the child was reported to have confusion, being overactive and over talkative for 3 days prior to attending the scheduled hematology clinic. Complete Blood Count showed a marked decrease in White blood cells. With the rapid patient’s clinical deterioration after initiation of HU, indicates that detailed medical investigations is crucial in disease management and the use of Tyrosine Kinase inhibitors should be encouraged as it has shown to be more tolerated and better survival outcomes in treatment of CML.