Ann Thorac Surg 2007;83:1188-1190
© 2007 The Society of Thoracic Surgeons
Case Reports
Unusual Case of Hemoptysis 13 Years After Bilobectomy
Subroto Paul, MD,
Siva Raja, MD, PhD,
Stacey Su, MD,
Jacques P. Fontaine, MD,
Lambros Zellos, MD,
David J. Sugarbaker, MD*
Division of Thoracic Surgery, Department of Surgery, Brigham & Women’s Hospital, Boston, Massachusetts
Accepted for publication September 27, 2006.
* Address correspondence to Dr Sugarbaker, Brigham and Women’s Hospital, Division of Thoracic Surgery, 75 Francis St, Boston, MA 02115 (Email: dsugarbaker{at}partners.org).
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Abstract
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We describe a 68-year-old man with recurrent episodes of hemoptysis. The patient had undergone right upper and middle lobectomies for a benign cystic lesion 13 years ago and coronary artery bypass grafting for ischemic heart disease immediately before the onset of hemoptysis. Diagnostic work-up revealed that pulmonary arterial flow to the remaining right lower lobe had been inadvertently compromised by prior ligation of the right main pulmonary artery during the time of his bilobectomy, with the development of a robust collateral blood supply derived from bronchial and intercostals arteries. Successful treatment required percutaneous embolization of the dominant bronchial collaterals, followed by completion pneumonectomy.
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Introduction
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Hemoptysis can result from a variety of causes. Bronchial arteries, although responsible for less blood flow to the lung than the pulmonary arteries, provide most of the blood supply to the airways and, hence, are the source behind many cases of hemoptysis. Any pathologic process that leads to erosion into the bronchial arteries or increases blood flow to the bronchial arteries can lead to hemoptysis, as described in this case.
A 68-year-old man began experiencing recurrent episodes of hemoptysis after undergoing coronary artery bypass grafting (CABG) for ischemic heart disease. His episodes began 2 weeks after his CABG procedure and consisted of coughing up 50 to 100 mL of blood on each occasion. Thirteen years prior, the patient had undergone right upper and middle lobectomies for a right upper lobe mass at an outside institution. Pathology showed this to be a benign cystic lesion with adjacent bronchiectasis and emphysematous changes. His other medical comorbidities included hypertension, hypercholesterolemia, and type 2 diabetes mellitus.
The patient’s post-CABG regimen of clopidogrel and warfarin was promptly discontinued. A chest roentgenogram was unrevealing, except for slight volume loss in the right lung hemothorax. Further diagnostic work-up with a chest computed tomography scan revealed that there was no pulmonary arterial flow to the remaining right lower lobe (Fig 1A), and a ventilation-perfusion scan confirmed perfusion only to the left lung. A bronchial arteriogram showed dilated, tortuous right intercostal and bronchial arteries to the right lower lobe, with venous drainage through the inferior pulmonary vein (Fig 1B). The left-sided vessels were of normal caliber (Fig 1B).
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Fig 1. (A) Chest computed tomography scan with intravenous contrast demonstrates a cutoff of flow in the right proximal pulmonary artery. (B) Angiogram demonstrates robust collateral intercostal and bronchial arterial flow. (C) Representative preembolization and (D) postembolization angiograms demonstrate embolization of right intercostal and associated bronchial vessel.
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The patient initially underwent embolization of the dominant bronchial arteries. His hemoptysis persisted, and he was referred to our institution. Preoperatively, he underwent repeat angiogram and embolization of six intercostal arteries perfusing the remaining right lower lobe (Fig 1C, 1D). Additional diagnostic workup consisted of pulmonary function tests (forced expiratory volume in 1 second of 1.60 liters, 68% predicted; forced vital capacity of 2.16 L, 73% predicted) and an echocardiogram (ejection fraction, 0.55; pulmonary artery systolic pressures of 54 mm Hg plus right atrial pressure).
The patient was taken to the operating room for a reoperative right thoracotomy. Initial exploration revealed multiple dense adhesions with large collateral vessels. The hilum was controlled, and the pulmonary venous drainage to the right lower lobe was divided. The adhesions were then taken down with cautery. The bronchus was divided, and the bronchial stump was buttressed with a pericardial fat pad. The thoracotomy was then closed once hemostasis was achieved.
The patient had an uneventful postoperative course and was discharged home on postoperative day 5. Pathology showed lung parenchyma with prominent bronchial arteries, peripheral capillary varicosities, and focal alveolar hemorrhage.
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Comment
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This report describes a case of pulmonary artery ligation leading to the delayed presentation of hemoptysis more than a decade postoperatively. The unintentional ligation of pulmonary arteries is a very rare but documented complication in thoracic surgery. More commonly, this phenomenon has been reported in the pediatric population undergoing patent ductus arteriosus closure where the left pulmonary artery was inadvertently ligated [1–3]. In these patients, asymmetric pulmonary blood flow is noted on chest roentgenography with the development of pulmonary hypertension. It is of interest that there are reports of the reconstitution of pulmonary arterial blood flow in some of these patients, including one at 6 months’ postligation [1–3].
We have presented a rarely reported case of pulmonary artery ligation in the setting of a lobectomy. The original operative report for this patient noted that an "aberrant third pulmonary vein that drains directly into the superior vena cava" was ligated. This may have been the misidentified right main pulmonary artery. The patient’s postoperative course after his initial bilobectomy was unremarkable, and the diagnosis was not suspected from the chest roentgenograms. The patient’s indolent course and delayed presentation of hemoptysis more than a decade later is a result of the development of pulmonary hypertension and the subsequent gradual development of collateral bronchial circulation to the remaining lower lobe. There was no frank infarction of the lung at the time of his initial bilobectomy as may be expected owing to the presence of the bronchial circulation. In this case, the bronchial circulation may have been robust to begin with given the patient’s underlying bronchiectasis.
The presentation of hemoptysis is not unexpected given the increased collateral circulation to the right lung and the accompanying pulmonary hypertension. Nonetheless, hemoptysis is an uncommon manifestation of unilaterally absent pulmonary artery, whether acquired or congenital [4–6]. It remains unclear what triggered the patient’s hemoptysis after CABG. It may have been the anticoagulation postoperatively or a transient decrease in ventricular function after cardiopulmonary bypass. Regardless, the systemic blood flow from the bronchial circulation and the resulting capillary turgidity resulted in endobronchial and intraalveolar hemorrhage. Other symptoms may include exertional dyspnea and angina from the underlying pulmonary hypertension, which in our case was likely caused by the patient’s underlying coronary disease.
The management of these patients requires a systematic approach. Bronchoscopy must be initially done to rule out any endobronchial lesion and to localize the bleeding site. Percutaneous embolization of bronchial collateral vessels is another option. In our setting, however, embolization alone was not a definitive treatment owing to the presence of an enriched collateral system. Embolization would only result in new collaterals developing and the eventual recannulization of those embolized. It does, however, reduce blood flow to the remaining lung parenchyma and thereby reduces intraoperative hemorrhage, which can be significant from the collateralized adhesions and lung parenchyma. Other surgical options include restoring blood flow by reestablishing patency as has been done in the aforementioned pediatric cases. Obviously in our case, this was not possible nor desired 13 years after bilobectomy, with collaterals already formed. Completion pneumonectomy, in our case, was the best option as it eliminated the source of hemoptysis—the bronchial collaterals. No other treatment plan offers this definitive solution.
In conclusion, the ligation of the pulmonary artery during a lobectomy is a rarely reported complication in thoracic surgery. In performing a completion pneumonectomy in these patients, angiography with embolization is a necessary tool in reducing intraoperative blood loss.
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References
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- Jaffe RB, Orsmond GS, Veasy LG. Inadvertent ligation of the left pulmonary artery Radiology 1986;161:355-357.[Abstract/Free Full Text]
- Orzel JA, Monaco MP. Inadvertent ligation of the left pulmonary artery instead of patent ductus arteriosusNoninvasive diagnosis by pulmonary perfusion imaging. Clin Nucl Med 1986;11:629-631.[Medline]
- Pontius RG, Danielson GK, Noonan JA, Judson JP. Illusions leading to surgical closure of the distal left pulmonary artery instead of the ductus arteriosus J Thorac Cardiovasc Surg 1981;82:107-113.[Abstract]
- Ten Harkel ADJ, Blom NA, Ottenkamp J. Isolated unilateral absence of a pulmonary artery Chest 2002;122:1471-14774.[Medline]
- Thomas P, Reynaud-Gaubert M, Bartoli JM, et al. Exsanguinating hemoptysis revealing the absence of left pulmonary artery in an adult Ann Thorac Surg 2001;72:1748-1750.[Abstract/Free Full Text]
- Farghly E, Bousamra M. Hemoptysis resulting from unilateral pulmonary artery agenesis Ann Thorac Surg 2002;74:255-257.[Abstract/Free Full Text]
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Abstract
Introduction
