- Email: [email protected]
Postoperative pulmonary arteriovenous fistula resulting in recurrent cryptogenic stroke
International Journal of Cardiology 117 (2007) e43 – e45 www.elsevier.com/locate/ijcard
Letter to the Editor
Postoperative pulmonary arteriovenous fistula resulting in recurrent cryptogenic stroke Suzanne Fateh-Moghadam ⁎, Rainer Dietz, Wolfgang Bocksch Department of Cardiology, Universitaetsmedizin Charite-Campus Virchow, Humboldt Universitaet zu Berlin, Augustenburger Platz 1, 13353 Berlin, Germany Received 19 August 2006; accepted 2 November 2006 Available online 2 January 2007
Abstract We report on a 66-year-old patient with a history of two cryptogenic strokes and a patent foramen ovale (PFO) who received a transcatheter closure of his PFO in our institution, but shortly after the intervention there was still a relevant right-to-left shunt. The following work-up showed an isolated pulmonary arteriovenous fistula as the real cause for the right-to-left shunt and hence the two strokes. © 2006 Elsevier Ireland Ltd. All rights reserved. Keywords: Pulmonary arteriovenous fistula; Cryptogenic stroke; Transcatheter closure of a persistent foramen ovale; Contrast echocardiography
A 66-year-old man with a history of recurrent strokes and a patent foramen ovale (PFO) was scheduled for operative closure of the PFO at another institution. He consulted our clinic for a second opinion regarding the feasibility of transcatheter closure. Other causes for the cerebrovascular events such as obstructive atherosclerosis of the extracranial arteries, left heart thrombus and intermittent atrial fibrillation were excluded. Hypercoagulability was absent as the laboratory work up showed no APC resistance, no protein C or S deficiency, the absence of antiphospholipid antibodies, and no prothrombin gene mutation G20210A. The external contrast transesophageal echocardiogram (TEE) revealed a small PFO with a spontaneous interatrial right-to-left shunt, suggesting paradoxical embolism as the most probable cause of the strokes, even though no coincident deep venous thrombi were documented. The patient's past medical history consisted of a Billroth I and II operation 1964/1965. Before the first stroke, an indurated bronchiectasis was treated by wedge resection of the ⁎ Corresponding author. E-mail address: [email protected] (S. Fateh-Moghadam). 0167-5273/$ - see front matter © 2006 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijcard.2006.11.033
left lower lobe in 2000, and a well differentiated papillary adenocarcinoma of the left lower lobe was resected in 2001. Postoperatively, he complaint of frequent headache and dizziness. After the first ischemic stroke in 2001 (left posterior cerebral artery territory), he was put on dual antiplatelet therapy (Aspirin 100 mg/d, Clopidogrel 75 mg/d). In 2004, he suffered another ischemic stroke (right anterior cerebral artery territory). On admission, physical examination was normal up to a decreased inspiration sound over the left lower lobe of the lung and an abdominal scar resulting from the Billroth I and II operation. No residual neurologic deficit was present. We recommended a transcatheter closure of the PFO and performed the procedure successfully under flouroscopic guidance with a 25-mm-Amplatzer PFO Occluder (AGA Medical, Golden Valley, MN, USA). Immediately after the procedure, we confirmed the correct position of the occluder by contrast transthoracic echocardiography (TTE), but a significant large residual right-to-left shunt (Fig. 1A) was still present, entering the left atrium at the origin of the left lower pulmonary vein, suggesting pulmonary arteriovenous fistula. Selective digital subtraction angiography of the left pulmonary artery revealed a medium sized pulmonary arteriovenous fistula (Fig. 2A) arising from the left lower lobe branch
e44
S. Fateh-Moghadam et al. / International Journal of Cardiology 117 (2007) e43–e45
artery and draining to the left lower pulmonary vein. Embolization and complete closure of the fistula were performed with implantation of 3 detachable coils (William Cook Europe, Bjaeverskov, Denmark, Fig. 2B). After this procedure contrast TTE (Fig. 1B) demonstrated no residual right-to-left shunt. Four months later, complete closure could be confirmed by TTE, and the patient reported an improvement of his previous headache and dizziness. 1. Discussion
Fig. 1. (A) Transthoracic contrast echocardiography (TTE, apical 4chamber-view) after PFO closure showing a residual right-to-left shunt, entering the left atrium at the origin of the left lower pulmonary vein →, suggesting pulmonary arteriovenous fistula. (B) After embolization of the pulmonary arteriovenous fistula, showing no residual right-to-left shunt.
This case clearly demonstrates that a pulmonary arteriovenous fistula should always be taken into account in patients with cryptogenic stroke, even if a PFO is present [1]. Contrast echocardiography [2] hereby is an extremely useful tool for diagnosis. The technique involves injection of echo contrast into a peripheral vein. The appearance of bubbles in the left atrium confirms right-to-left shunting and if they can be seen entering the left atrium via a single pulmonary vein, contrast echocardiography confirms the exact anatomic localization of the fistula [2]. For this reason it is always necessary to monitor the pulmonary veins during injection of the contrast by TEE. Of course, the small PFO could have been the cause for the recurrent strokes in this patient, however, respecting the history with onset of symptoms after thoracic surgery, suggests strongly, that the fistula is the most likely cause for the presumed paradoxical embolisms. We have to suspect, that the fistula had been originated secondarily from the surgical interventions which is a very rare, but previously described complication after thoracic surgery [3]. Depending on size, almost all pulmonary arteriovenous fistulas [4,5] can be closed percutaneously as it was done in our patient. Although PFO closure was not primarily indicated, a predisposition for paradoxical embolism seems to exist in this patient.
Fig. 2. (A) Selective digital subtraction angiography (DSA). Selective injection into the left lower pulmonary artery shows the arterial supply (p) and venous drainage (Y) of the pulmonary arteriovenous fistula. (B) Selective injection into the left lower pulmonary artery following successful coil embolization of the feeding artery (8/5 mm, 2x 5/5 mm).
S. Fateh-Moghadam et al. / International Journal of Cardiology 117 (2007) e43–e45
Whether an open heart surgery would have led to the detection and closure of the fistula is questionable. References [1] Peters B, Ewert P, Schubert S, Abdul-Khaliq H, Lange PE. Rare case of pulmonary arteriovenous fistula simulating residual defect after transcatheter closure of patent ovale for recurrent paradoxical embolism. Catheter Cardiovasc Interv 2005;64:348–51. [2] Chessa M, Drago M, Krantunkov P, et al. Differential diagnosis between patent foramen ovale and pulmonary arteriovenous fistula in two
e45
patients with previous cryptogenic stroke caused by presumed paradoxical embolism. J Am Soc Echocardiogr 2002;15:845–6. [3] Riehl G, Chaffanjon P, Frey G, Sessa C, Brichon PY. Postoperative systemic artery to pulmonary vessel fistula: analysis of three cases. Ann Thorac Surg 2003;76:1873–7. [4] Pick A, Deschamps C, Stanson AW. Pulmonary arteriovenous fistula: presentation, diagnosis, and treatment. World J Surg 1999;23:1118–22. [5] Zanchetta M, Rigatelli G, Pedon L, Zennaro M, Maiolino P, Onorato E. Transcatheter Amplatzer duct occluder closure of direct pulmonary to left atrium communication. Catheter Cardiovasc Interv 2003;58:107–10.
Letter to the Editor
Postoperative pulmonary arteriovenous fistula resulting in recurrent cryptogenic stroke Suzanne Fateh-Moghadam ⁎, Rainer Dietz, Wolfgang Bocksch Department of Cardiology, Universitaetsmedizin Charite-Campus Virchow, Humboldt Universitaet zu Berlin, Augustenburger Platz 1, 13353 Berlin, Germany Received 19 August 2006; accepted 2 November 2006 Available online 2 January 2007
Abstract We report on a 66-year-old patient with a history of two cryptogenic strokes and a patent foramen ovale (PFO) who received a transcatheter closure of his PFO in our institution, but shortly after the intervention there was still a relevant right-to-left shunt. The following work-up showed an isolated pulmonary arteriovenous fistula as the real cause for the right-to-left shunt and hence the two strokes. © 2006 Elsevier Ireland Ltd. All rights reserved. Keywords: Pulmonary arteriovenous fistula; Cryptogenic stroke; Transcatheter closure of a persistent foramen ovale; Contrast echocardiography
A 66-year-old man with a history of recurrent strokes and a patent foramen ovale (PFO) was scheduled for operative closure of the PFO at another institution. He consulted our clinic for a second opinion regarding the feasibility of transcatheter closure. Other causes for the cerebrovascular events such as obstructive atherosclerosis of the extracranial arteries, left heart thrombus and intermittent atrial fibrillation were excluded. Hypercoagulability was absent as the laboratory work up showed no APC resistance, no protein C or S deficiency, the absence of antiphospholipid antibodies, and no prothrombin gene mutation G20210A. The external contrast transesophageal echocardiogram (TEE) revealed a small PFO with a spontaneous interatrial right-to-left shunt, suggesting paradoxical embolism as the most probable cause of the strokes, even though no coincident deep venous thrombi were documented. The patient's past medical history consisted of a Billroth I and II operation 1964/1965. Before the first stroke, an indurated bronchiectasis was treated by wedge resection of the ⁎ Corresponding author. E-mail address: [email protected] (S. Fateh-Moghadam). 0167-5273/$ - see front matter © 2006 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijcard.2006.11.033
left lower lobe in 2000, and a well differentiated papillary adenocarcinoma of the left lower lobe was resected in 2001. Postoperatively, he complaint of frequent headache and dizziness. After the first ischemic stroke in 2001 (left posterior cerebral artery territory), he was put on dual antiplatelet therapy (Aspirin 100 mg/d, Clopidogrel 75 mg/d). In 2004, he suffered another ischemic stroke (right anterior cerebral artery territory). On admission, physical examination was normal up to a decreased inspiration sound over the left lower lobe of the lung and an abdominal scar resulting from the Billroth I and II operation. No residual neurologic deficit was present. We recommended a transcatheter closure of the PFO and performed the procedure successfully under flouroscopic guidance with a 25-mm-Amplatzer PFO Occluder (AGA Medical, Golden Valley, MN, USA). Immediately after the procedure, we confirmed the correct position of the occluder by contrast transthoracic echocardiography (TTE), but a significant large residual right-to-left shunt (Fig. 1A) was still present, entering the left atrium at the origin of the left lower pulmonary vein, suggesting pulmonary arteriovenous fistula. Selective digital subtraction angiography of the left pulmonary artery revealed a medium sized pulmonary arteriovenous fistula (Fig. 2A) arising from the left lower lobe branch
e44
S. Fateh-Moghadam et al. / International Journal of Cardiology 117 (2007) e43–e45
artery and draining to the left lower pulmonary vein. Embolization and complete closure of the fistula were performed with implantation of 3 detachable coils (William Cook Europe, Bjaeverskov, Denmark, Fig. 2B). After this procedure contrast TTE (Fig. 1B) demonstrated no residual right-to-left shunt. Four months later, complete closure could be confirmed by TTE, and the patient reported an improvement of his previous headache and dizziness. 1. Discussion
Fig. 1. (A) Transthoracic contrast echocardiography (TTE, apical 4chamber-view) after PFO closure showing a residual right-to-left shunt, entering the left atrium at the origin of the left lower pulmonary vein →, suggesting pulmonary arteriovenous fistula. (B) After embolization of the pulmonary arteriovenous fistula, showing no residual right-to-left shunt.
This case clearly demonstrates that a pulmonary arteriovenous fistula should always be taken into account in patients with cryptogenic stroke, even if a PFO is present [1]. Contrast echocardiography [2] hereby is an extremely useful tool for diagnosis. The technique involves injection of echo contrast into a peripheral vein. The appearance of bubbles in the left atrium confirms right-to-left shunting and if they can be seen entering the left atrium via a single pulmonary vein, contrast echocardiography confirms the exact anatomic localization of the fistula [2]. For this reason it is always necessary to monitor the pulmonary veins during injection of the contrast by TEE. Of course, the small PFO could have been the cause for the recurrent strokes in this patient, however, respecting the history with onset of symptoms after thoracic surgery, suggests strongly, that the fistula is the most likely cause for the presumed paradoxical embolisms. We have to suspect, that the fistula had been originated secondarily from the surgical interventions which is a very rare, but previously described complication after thoracic surgery [3]. Depending on size, almost all pulmonary arteriovenous fistulas [4,5] can be closed percutaneously as it was done in our patient. Although PFO closure was not primarily indicated, a predisposition for paradoxical embolism seems to exist in this patient.
Fig. 2. (A) Selective digital subtraction angiography (DSA). Selective injection into the left lower pulmonary artery shows the arterial supply (p) and venous drainage (Y) of the pulmonary arteriovenous fistula. (B) Selective injection into the left lower pulmonary artery following successful coil embolization of the feeding artery (8/5 mm, 2x 5/5 mm).
S. Fateh-Moghadam et al. / International Journal of Cardiology 117 (2007) e43–e45
Whether an open heart surgery would have led to the detection and closure of the fistula is questionable. References [1] Peters B, Ewert P, Schubert S, Abdul-Khaliq H, Lange PE. Rare case of pulmonary arteriovenous fistula simulating residual defect after transcatheter closure of patent ovale for recurrent paradoxical embolism. Catheter Cardiovasc Interv 2005;64:348–51. [2] Chessa M, Drago M, Krantunkov P, et al. Differential diagnosis between patent foramen ovale and pulmonary arteriovenous fistula in two
e45
patients with previous cryptogenic stroke caused by presumed paradoxical embolism. J Am Soc Echocardiogr 2002;15:845–6. [3] Riehl G, Chaffanjon P, Frey G, Sessa C, Brichon PY. Postoperative systemic artery to pulmonary vessel fistula: analysis of three cases. Ann Thorac Surg 2003;76:1873–7. [4] Pick A, Deschamps C, Stanson AW. Pulmonary arteriovenous fistula: presentation, diagnosis, and treatment. World J Surg 1999;23:1118–22. [5] Zanchetta M, Rigatelli G, Pedon L, Zennaro M, Maiolino P, Onorato E. Transcatheter Amplatzer duct occluder closure of direct pulmonary to left atrium communication. Catheter Cardiovasc Interv 2003;58:107–10.