Neurogenic pulmonary edema (NPE) is defined as acute pulmonary edema develops after a significant central nervous system insult. Although NPE has been recognized for a long time, it is still underdiagnosed in clinical practice. The exact mechanism remains unclear, but the activation of sympathetic nervous system and a catecholamine surge play important roles. Common clinical presentations are dyspnea, hypoxia and pink frothy sputum. The mortality rate is high, but recovery is usually good in surviving patients. Here we report a 62-year-old woman presenting with abrupt onset respiratory distress during thrombolytic therapy after acute ischemic stroke involving the left insular and fronto-temporo-parietal lobes. Diagnosis of NPE following acute ischemic stroke was made. Review of literature showed that NPE following ischemic stroke is rare, with only few cases previously reported in the literature.