Introduction: The natural history of patients with arteriovenous malformations (AVMs) suggests that 10-17% die due to hemorrhage and 40-50% experience serious physical deterioration of their working capacity in their 20-40-year period after presentation and treatment. Deep basal ganglia and thalamus lesions account for 3-12% of all AVMs and some literature suggests that these lesions have a more hostile clinical course. Case Presentation: 16-year-old male, a student with no known chronic illnesses came to our hospital as a referral from Lubaga hospital. He presented with fifteen days’ history of sudden onset of severe headache with no known relieving or exacerbating factors. This was associated with episodes of vomiting, altered mentation, right sided weakness, however there was no history of convulsions, blurring of vision or trauma. This was the first time for this kind of presentation. On admission: He was lethargic, moderately dehydrated, not pale nor jaundiced. BP was 109/52mmHg, HR of 60bpm, SPO2=96% on room air. Glasgow Coma Scale score of 14/15, V=4, PEAL, soft neck, normal muscle bulky with a power of 2/5 on the left side and 4/5 on the right side. Successful surgical resection of the AVM was done at Mbarara regional referral Hospital-Uganda. Currently the patient is back in the community and has returned to school. Conclusion: Children who present with intracranial hemorrhage from a previously undiagnosed AVM have a 12% chance of sudden death. Clinical triggers of hemorrhage are unpredictable, and all fatal cases were in locations with high risk of potential herniation. These data support a proactive, aggressive approach toward definitive treatment of AVMs in children and adults. Patients with diagnosed AVM can once again get a chance to survive like other children in the community in LMICs like Uganda by undergoing open surgery, although advanced opportunities are still limited.