Case report

A 26-year-old man referred to our Emergency Department complaining motor aphasia in the morning (lasted about 1 hour) followed by migraine with aura. On arrival, he was eupneic, apiretic, with normal pulse oximetry (97%), regular heart rate (65 beats per minute), and bilateral blood pressure of 100/70 mmHg. Physical examination was normal. He was then admitted to the neurology ward for further diagnostic tests. Medical history collection revealed perinatal stroke, previous seizures, recurrent migraine, and a patent foramen ovale, diagnosed the year before by transesophageal echocardiography. Cranial CT scan and MRI were performed and revealed recent ischemic, embolic lesions in the left temporal and frontal cerebral lobe. Additional diagnostic tests were performed to exclude other causes of cerebral ischemia. Carotid Doppler, lower limbs venous ultrasound, electrocardiogram, 24-hour Holter monitoring and laboratory tests were normal.

The evidence of stroke in a patient with previously known patent foramen ovale strongly suggested paradoxical embolism. He was diagnosed with patent foramen ovale-related stroke and he was therefore planned for percutaneous closure of septal defect.

Pre-procedural contrast transoesophageal echocardiography (performed with a Vivid 7 Dimension ultrasound machine, General Electric Healthcare, Norfolk, VA, USA) showed a massive early right-to-left shunt through patent foramen ovale. Percutaneous closure of patent foramen ovale was performed using standardised techniques,^{Reference Meier5,Reference Diaz, Cubeddu and Rengifo-Moreno6} with transoesophageal echocardiography guidance. Balloon-sizing was avoided in order to prevent septal damage. An 18-mm Amplatzer patent foramen ovale occluder (St Jude Medical, Minneapolis, MN, USA) was implanted.

At the end of the procedure, contrast transoesophageal echocardiography (Fig 1A) revealed correct placement of the device with resolution of the early right-to-left shunt, though a persistent and significant late shunt was still evident, with left atrial opacification after five cardiac cycles following right atrial opacification. Since a pulmonary arteriovenous fistula was suspected, an agitated saline injection in the pulmonary trunk was performed, showing left atrial opacification. A CT angiogram was later performed and showed four pulmonary arteriovenous fistulas in the left lung inferior lobe (Fig 1C). The patient underwent percutaneous closure of three pulmonary arteriovenous fistulas (Fig 1B) with embolisation coils. The fourth pulmonary fistula was left untreated because of the inability to access the target site (located in a peripheral position in the superior lobe) and the small size of the lesion (<4 mm). At 6-month follow-up, the patient was asymptomatic, without further episodes of cerebral ischemia. The three pulmonary arteriovenous fistulas were completely closed on both pulmonary and CT angiogram (Fig 1 D/E), while contrast transoesophageal echocardiography showed a trivial residual shunt.

Figure 1.

Patent foramen ovale closure with pulmonary arteriovenous fistulas detection and treatment. (a) Intraoperative contrast transoesophageal echocardiography showing correct deployment of the occluder device (OD) but persistence of late shunt (MB, microbubbles) in the left atrium. (b/c) Pulmonary/CT angiogram showing one of the four pulmonary arteriovenous fistulas before its percutaneous closure. (d) Post-procedural pulmonary angiogram showing closed pulmonary arteriovenous fistulas. (e) Six-month CT angiogram showing good results of pulmonary arteriovenous fistulas closure over time.