RATIONALE: Computed tomography plays a key role in the initial evaluation of suspected acute stroke by ruling out the possibility of hemorrhage before thrombolysis. Recently, many reports have described cases of symptomatic microbleeds, and there may also have been a case of computed tomography- negative intracerebral hemorrhage. PATIENT CONCERNS: A 70-year-old female patient who had a history of lacunar infarction and severe small vessel disease developed dysarthria. On brain non-contrast computed tomography there was no evidence of intracerebral hemorrhage. However, brain magnetic resonance imaging performed at 3 hours after the initial computed tomography showed cerebral hemorrhage. DIAGNOSES: The diagnosis was computed tomography-negative intracerebral hemorrhage. INTERVENTIONS: The patient was treated with cilostazole 100 mg twice a day with blood pressure management. OUTCOMES: The dysarthria was fully recovered within 5 days and the patient did not suffer recurrent stroke symptoms over the following 2 years. LESSONS: In patients with underlying severe small vessel disease and microbleeds, there could be computed tomography-negative hemorrhage and susceptibility weighted magnetic resonance image could be needed. More attention is required before applying thrombolysis therapy because there is a possibility of cerebral hemorrhage in those patients.