Ann Thorac Surg 2005;80:731-733
© 2005 The Society of Thoracic Surgeons
Case report
Aortobronchial Fistula in a Pediatric Patient With Massive Hemoptysis: Treatment by Means of an Aortic Endograft
Rahul K. Khare, MD
a
,
Philip D. Settimi, MD
d
,
Nkechi I. Mba, MD
b
,
Daniel S. Wechsler, MD, PhD
b
,
Susan L. Bratton, MD, MPH
b
,
David M. Williams, MD
c
,
*
a Department of Emergency Medicine, University of Michigan, Ann Arbor, Michigan, USA
b Department of Pediatrics, University of Michigan, Ann Arbor, Michigan, USA
c Department of Radiology, University of Michigan, Ann Arbor, Michigan, USA
d Department of Medical School, University of Michigan, Ann Arbor, Michigan, USA
Accepted for publication February 10, 2004.
* Address reprint requests to Dr Williams, Department of Radiology, University Hospitals, B1-D530, Ann Arbor, MI48109-0030 (Email: davidwms{at}med.umich.edu).
 |
Abstract
|
|---|
We present an 11-year-old girl with acute myelogenous leukemia and hemoptysis from abscess erosion into the descending thoracic aorta. We report a pediatric case of an aortobronchial fistula treated with an aortic endograft and discuss the technical limitations and potential complications of this procedure.
|
Introduction
|
|---|
Massive hemoptysis secondary to an aortobronchial fistula (ABF) is a life-threatening emergency. Since the mid-1990s, minimally invasive endovascular stent graft, or endograft, techniques have been reliably used in select adult patients.
An 11-year-old girl presented to the emergency department with severe bruising and was admitted for acute myelogenous leukemia (AML), M2 subtype according to the FAB classification. She received two cycles of induction chemotherapy consisting of idarubicin/daunorubicin, dexamethasone, cytarabine, thioguanine, and etoposide. Shortly after the second cycle of chemotherapy, while neutropenic, she developed a left lower lobe pneumonia and abscess. Computed tomography-guided aspiration demonstrated Aspergillus bacteria (Fig 1A).
View larger version (112K):
[in this window]
[in a new window]
|
Fig 1. Serial computed tomographic (CT) scans show evolution of the juxtaaortic lung abscess. At initial CT examination, a fluid density with a contrast-enhancing rim is demonstrated (A). A later CT scan at the same level shows the aortobronchial fistula project- ing from the aorta at the 5 o’clock position (B). After placement of the endograft and lung debridement, a CT scan at the time of dis- charge shows residual extrapleural soft-tissue density, concave outward, but no contrast leak (C).
|
|
Antifungal treatment with amphotericin B and caspofungin was started. Seven days later, severe intrascapular back pain and massive hemoptysis (approximately 1 L) developed. An emergent computed tomographic scan of the thorax and aortic angiography revealed a left lung abscess 8 cm in diameter with an ABF (Fig 1B). The fistula came directly off the descending thoracic aorta (Fig 2A). Because she was a poor surgical candidate, noninvasive treatment was planned. Embolization of the fistula with Tornado microcoils (Cook Incorporated, Bloomington, IN) achieved transient hemodynamic stability. However, 3 hours later, she developed recurrent hemoptysis and hypotension.
View larger version (165K):
[in this window]
[in a new window]
|
Fig 2. Initial aortography in the left anterior oblique projection shows a nipple-like aortobronchial fistula (arrow) arising from the proximal descending aorta (A). Aortography after endograft placement shows that the fistula has been excluded from the aortic lumen (B).
|
|
The patient received a transfusion of 4 U of packed red blood cells and was returned to angiography for a planned deployment of an endograft. The right femoral artery was exposed but was too small to allow passage of the endograft delivery system and surgical exposure of the common iliac was required. Two overlapping AneuRx endografts (Medtronic AVE; Santa Rosa, CA), 20 mm in diameter and 3.75 cm long, were deployed under fluoroscopic guidance, beginning approximately 2 cm distal to the left subclavian artery (Fig 2B). The endograft was dilated using a balloon 18 mm in diameter and 40 mm long. The hemoptysis resolved.
Because the abscess did not resolve, she later underwent a left lower lobectomy and evacuation of hematoma and embolization coils 11 days later. She tolerated the operation well and was discharged on postlobectomy day 6 in good condition. At the time of discharge, computed tomography showed a small periaortic soft-tissue density but no leak (Fig 1C). She continued antifungal therapy for 5 months and tolerated three additional cycles of intensive chemotherapy. She remains in remission and free of infection and recurrent hemoptysis 18 months later.
|
Comment
|
|---|
Patients with leukemia and lymphoma who are undergoing cytotoxic chemotherapy are predisposed to invasive fungal infections [1, 2]. In particular, invasive aspergillosis is associated with a poor prognosis. Fungal invasion of blood vessels with severe hemoptysis has been reported in fatal cases [3] and is an ominous sign that warrants aggressive therapy. Although embolization of a pseudoaneurysm with stainless steel coils would not be expected to provide a durable cure, it did arrest the ongoing hemorrhage in this patient and allow temporary hemodynamic resuscitation. Placing an endograft adjacent to an abscess presents a theoretical risk of colonization of the fabric and inability to clear the primary infection. The use of endografts to treat infected aortobronchial fistulas has been reported to provide long-term relief in adults [4, 5], but their use in children requires careful consideration.
Delivery of the endografts in children is limited by the device size. The delivery catheter for the AneuRx (Medtronic AVE) device is 7 mm in diameter and accommodates endografts as large as 28 mm in diameter. Although the average diameter of the femoral artery in a 13-year-old child is 10.4 mm [6], our patient was smaller and required cannulation of the common iliac artery. If the device could not have been delivered from the femoral or iliac artery, then deployment through an aortotomy remote from the target lesion would be required.
Another limitation to endograft use in children is the need to accommodate future aortic growth. The AneuRx (Medtronic AVE) endograft is a self-expanding device that adheres to the aortic wall by friction. Consequently, the device is selected to be "oversized." At the time of procedure, the descending aorta measured 18 mm in diameter at the level of the aortic isthmus and 16 mm distally. A 22-mm diameter device was preferred, but none was available. A 20-mm diameter device provided immediate hemostasis. In a normal woman, the aortic diameter at the level of the isthmus, varies from 21 mm at 21 to 40 years of age to 29 mm at older than 61 years of age [7]. Given this potential diameter mismatch, the endograft may cause a coarctation-like stenosis as the aorta grows, assuming that it adheres to the vessel wall. However, the endograft may not incorporate into the aortic wall, and as the aorta grows and exceeds the unconstrained diameter of the endograft, the device may migrate.
Finally, the structural durability of these devices has not been established. A clinical trial of a different endograft specifically designed to treat thoracic aortic aneurysms was recently suspended by the manufacturer because of component fractures detected in imaging follow-up in several patients. Because of these considerations, our patient will need long-term monitoring of extremity blood pressures to detect stenosis as well as serial roentgenograms to monitor the structural integrity and stability of the device.
In addition to demonstrating the effectiveness of an aortic endograft to resolve the acute bleeding episode, this case indicates that this procedure may be tolerated in a neutropenic leukemia patient at exceedingly high risk of disseminated infection. Aggressive treatment with antifungal therapy, together with these surgical approaches, permitted this patient to complete the prescribed chemotherapy course and be in remission 18 months later.
This case demonstrates the beneficial application of an unconventional, high-risk intervention in a child with acute leukemia. Endografts can be placed in the aorta of an unstable child in whom the risk and benefits of device placement are carefully considered and appear favorable compared with surgical intervention.
|
References
|
|---|
- Batra PS, Holinger LD. Etiology and management of pediatric hemoptysis Arch Otolaryngol Head Neck Surg 2001;127(4):377-382.[Abstract/Free Full Text]
- Kitabayashi A, Hirokawa M, Yamaguchi A, et al. Invasive pulmonary mucormycosis with rupture of the thoracic aorta Am J Hematol 1998;58:326-329.[Medline]
- Panos RJ, Barr LF, Walsh TJ, et al. Factors associated with fatal hemoptysis in cancer patients Chest 1988;94:1008-1013.[Abstract/Free Full Text]
- Leobon B, Roux D, Mugniot A, et al. Endovascular treatment of thoracic aortic fistulas Ann Thorac Surg 2002;74:247-249.[Abstract/Free Full Text]
- Kramer S, Pamler R, Seifarth H, et al. Endovascular grafting of traumatic aortic aneurysms in contaminated fields J Endovasc Ther 2001;8:262-267.[Medline]
- Steinberg C, Weinstock DJ, Gold JP, et al. Measurements of central blood vessels in infants and childrennormal values. Cathet Cardiovasc Diagn 1992;27:197-201.[Medline]
- Aronberg DJ, Glazer HS, Madsen K, et al. Normal thoracic aortic diameters by computed tomography J Comput Assist Tomogr 1984;8:247-250.[Medline]
This article has been cited by other articles:
|
|
|
|
 
G. H. Wheatley III, A. Nunez, O. Preventza, V. G. Ramaiah, J. A. Rodriguez-Lopez, J. Williams, D. Olsen, and E. B. Diethrich
Have we gone too far? Endovascular stent-graft repair of aortobronchial fistulas
J. Thorac. Cardiovasc. Surg.,
May 1, 2007;
133(5):
1277 - 1285.
[Abstract]
[Full Text]
[PDF]
|
|
|
Top
Abstract
Introduction
