Pulmonary arterial venous malformation (PAVM) is a risk factor for paradoxical embolism (PE) and cryptogenic stroke (CS).

CS caused by PE through PAVM with limb weakness as the first symptom is relatively rare in the clinic, and PAVM should be screened as an etiology in stroke patients without any other risk factors.

Transcranial Doppler ultrasound with bubble study (TCD-b) is non-invasive and sensitive in detecting right-to-left shunts; if TCD-b is positive, transesophageal echocardiography or transthoracic echocardiography should be conducted to distinguish between intra- and extracardiac shunts.

Computerized tomography angiogram or digital subtraction angiography of the pulmonary artery is the recommended gold standard for diagnosing and locating a PAVM.

Our case indicated that thrombolytic therapy within the time window combined with transcatheter embolotherapy and rehabilitation training may be a benefit for the recovery of patients with CS caused by PE resulting from PAVM.