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Ann Thorac Surg 1997;63:839-841
© 1997 The Society of Thoracic Surgeons

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Case Reports

Chronic Pain of Vascular Origin Caused by a Parietopulmonary Fistula of the Thoracic Wall

Service de Pneumologie et Service de Chirurgie Cardio-Vasculaire et Thoracique, Centre Hospitalier et Universitaire, Angers, France

Accepted for publication October 11, 1996.

	Abstract

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We describe the case history of a patient who suffered from dorsal thoracic nonpenetrating trauma after a fall, with rupture of the aortic isthmus treated by thoracotomy. Arteriovenous fistula between the thoracic wall and the lung, with hypervascularization of the left thoracic wall, was revealed by atypical chronic thoracic pain and a murmur. This case demonstrates that chronic thoracic pain of vascular origin must be considered.

	Introduction

Top Footnotes Abstract Introduction Comment References

 
After penetrating or nonpenetrating thoracic injury, thoracic lesions can be detected immediately or during the days or weeks following injury. However, in a few rare cases, lesions may appear some time after the injury took place. Posttraumatic pulmonary arteriovenous fistulas are among these late-onset complications [1].

In July 1992, after a fall from a height of 16 m, a 27-year-old man was admitted for fractures of the posterior arches of the 8th, 9th, 10th, and 11th right ribs and the 6th, 7th, and 8th left ribs. Subcutaneous right-sided thoracic emphysema was also present, but there was no pneumothorax. Rupture of the left diaphragm dome had occurred, and the stomach and the spleen were inside the left thorax. Injuries to the diaphragm and the spleen were such that surgical repair and splenectomy were required. The lung was normal, and blood from a spleen injury was evacuated. Considering the height of the fall and the apparent damages done, routine aortography was performed. A traumatically induced aneurysm of the aortic isthmus was found. Left-sided thoracotomy was performed in the 5th rib space. Extensive intrathoracic bleeding followed this operation, and rapid clotting prevented sufficient draining via the two drains inserted. A second operation was performed to aspirate this left-sided hemothorax, and recovery was then uncomplicated. During the two latter operations, no abnormality was observed.

During the following weeks, the patient complained of atypical left-sided thoracic pain radiating toward the front of the lower left thoracic wall, two rib spaces below the thoracotomy scar. A year after injury had taken place, the pain was always present and had not changed over time. The chronic pain was diagnosed as being caused by esophageal reflux, confirmed using pH measurements. The patient was treated medically for a period of 6 months, without any improvement, then an operation was performed: a posterior Toupet valve was inserted, and the columns were closed off. The patient continued to experience pain, however, which became constant.

Two years after the accident, a continuous murmur was detected in the area where the patient was experiencing pain. Aortography showed that the shape of the aorta was normal, particularly in the isthmus area, but that the left internal thoracic artery was dilated. On selective arteriography, one of the left internal thoracic artery branches was anastomosed with a pulmonary artery branch in the lingular region (Fig 1).

View larger version (138K): [in this window] [in a new window]   Fig 1. . (A) Left internal thoracic artery angiogram before embolization, on a posteroanterior projection. (B) Schematic representation of the internal thoracic artery-to-pulmonary arteriovenous fistula based on the angiogram: ( 1) left internal thoracic artery, (2) hypervascularization down the left internal thoracic artery, (3) vascular fistula, (4) pulmonary artery, (5) left inferior pulmonary vein, and (6) left atrium.

This disabling pain, which the patient had been experiencing for a period of 2 years, disappeared within 1 day. Two months later, there was no recurrence of pain and no abnormal revascularization of these lesions.

	Comment

Top Footnotes Abstract Introduction Comment References

 
This unusual case history raises two types of question. What are the mechanisms involved in the development of this fistula? What are the physiopathologic mechanisms causing pain?

Several theories can be used to explain the development of the vascular fistula between the circulatory system of the thoracic wall and the pulmonary circulation. First, one can consider that the fistula was present before the injury occurred: congenital fistulas located between the internal thoracic artery and a pulmonary artery have been described in the literature [2–6]. Acquired fistulas such as those appearing after tuberculosis [7], pleurisy [8], or pneumonia [9] can be also be considered. However, in both of these cases, during thoracotomy the surgeon would have noticed adhesions between the lung and the thoracic wall [2, 5, 8, 9], and this was not the case in this patient. Another possibility is that, during the two thoracic operations performed, an iatrogenic wound was induced and this in turn could have caused painful inflammation at the thoracic wall (due to hemothorax) and pulmonary parenchyma levels and subsequent fistula formation. The second theory is the usual one: that is, penetrating injury that has damaged the two vessels involved. In the case of this patient, the injury was not penetrating. In the third theory, it is considered that an indirect complication resulting from the initial dorsal injury (such as tearing of the visceral pleura caused by a blast mechanism) may be present. This process would have made it possible for the underlying pulmonary parenchyma to connect with the thoracic wall after an inflammatory process induced by the hemothorax.

The cause of pain is related to the presence of hypervascularization or the fistula, given that pain disappeared after embolization. The literature contains some references of 2 patients suffering from fistulas and thoracic pain [4, 5]. In 1 case [5], dull precordial pain was present, and in the other case [4], atypical thoracic pain was present. In these 2 cases, no hypervascularization was found. Atypical thoracic pain can also be a sign of an extrathoracic fistula [10]. On examination of the literature, it would thus appear unlikely that pain was caused by hypervascularization in this patient, even if the mechanism causing fistula-related pain has yet to be elucidated.

In this case, the nonpenetrating trauma and the observations during surgical interventions did not lead us to suggest a relation between the pain and a lesion. A computed tomographic scan or magnetic resonance imaging would have had to be made because the pain seemed to have a parietal origin, but the physical examination was always normal. Some authors have reported the use of radionuclide angiocardiography to reveal systemic–pulmonary fistula, the location of which was made by computed tomographic scan [7], but the patient had symptomatic high-output cardiac failure. Some authors have described the use of duplex and color Doppler ultrasonography to diagnose an iatrogenic intercostal arteriovenous fistula [11].

This case demonstrates that clinical examination must be performed at regular intervals where posttraumatic chronic pain is present. A vascular cause must always be considered. Vascular exploration of the painful area by Doppler ultrasonography and computed tomographic scan therefore must be performed when all other usual causative factors for pain have been eliminated, according to the patient's injuries and past history.

	Footnotes

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Address reprint requests to Dr Jeanfaivre, Service de Pneumologie, Centre Hospitalier et Universitaire, 49033 Angers Cedex 01, France.

	References

Top Footnotes Abstract Introduction Comment References

 

1. Kerr A, Sauter D. Acquired traumatic pulmonary arteriovenous fistula: case report. J Trauma 1993;35:484–6.[Medline]
2. Brain R, Kauntze R. Systemic–pulmonary arteriovenous aneurysm of chest wall and lung. Guy's Hosp Rep 1960;109:110–4.
3. Voll A, Marstrander F, Wexels P. Systemic–pulmonary shunt. Dis Chest 1964;45:396–401.
4. Kiphart RJ, Mackenzie JW, Templeton AW, Martin RA. Systemic–pulmonary arteriovenous fistula of the chest wall and the lung. J Thorac Cardiovasc Surg 1967;54:113–20.[Medline]
5. Stafford RW, Kronenberg MW, Dunbar JD, Wooley CF. Continuous precordial murmurs due to internal mammary artery fistulas. Am J Cardiol 1969;24:414–9.[Medline]
6. Robinson LA, Sabiston DC. Syndrome of congenital internal mammary-to-pulmonary arteriovenous fistula associated with mitral valve prolapse. Arch Surg 1981;116:1265–73.[Abstract/Free Full Text]
7. Chino M, Kawaguchi T, Sakai T, Okuno T. Intercostal- to-pulmonary arterial anastomosis, complicated by high-output heart failure: case report. Angiology 1991;42:256–60.
8. Burchell HB, Clagett OT. The clinical syndrome associated with pulmonary arteriovenous fistulas, including a case report of a surgical cure. Am Heart J 1947;34:151–62.
9. Prutzman LD, Flick JB. Pulmonary arteriovenous fistula with extensive thoracic wall collateral circulation. Bull Ayer Clin Lab 1954;21:23–9.
10. Marx M, Geller S, Bettman M. Traumatic aortocaval fistula presenting as atypical chest pain. Cardiovasc Intervent Radiol 1986;9:17–8.[Medline]
11. Derdeyn CP, Middleton WD, Allen BT, Nordlicht SM. Acquired intercostal arteriovenous fistula: color Doppler ultrasonographic diagnosis. J Ultrasound Med 1993;12: 679–81.[Medline]

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This Article Abstract Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Jeanfaivre, T. Articles by Enon, B. Search for Related Content PubMed PubMed Citation Articles by Jeanfaivre, T. Articles by Enon, B.