(Chest. 2002;121:988-990.)
© 2002
American College of Chest Physicians
Management of an Intrabronchial Rupture of Right Main Pulmonary Artery*
A Case Report
Christophe Pinet, MD;
Wojciech Palka, MD;
Dominique Metras, MD;
Pascal Thomas, MD;
Bernard Méric, MD and
Jean-François Dumon, MD, FCCP
*
From the Département des Maladies Respiratoires et Center Laser (Drs. Pinet, Palka, Méric, and Dumon) and the Service de Chirurgie thoracique (Dr. Thomas), Hôpital Sainte-Marguerite, Marseilles, France; the Service de Chirurgie Thoracique et Cardio-Vasculaire (Dr. Metras), Hôpital de la Timone, Marseilles, France.
Correspondence to: Christophe Pinet, MD, Département des Maladies Respiratoires, Hôpital Sainte-Marguerite, 270 Blvd Sainte-Marguerite, BP 29–13274 Marseille Cedex 09, France; e-mail: CPINET{at}mail.ap-hm.fr
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Abstract
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We report a case of massive endobronchial hemorrhage following a fistula formation of the right pulmonary artery to the right mainstem bronchus in a 15-year-old girl. The fistula had occurred 39 days after the patient had undergone bilateral lung transplantation for cystic fibrosis. The post-transplantation course was remarkable for bronchial colonization by Aspergillus at the site of right bronchial anastomosis and an episode of spontaneous, self-limited hemoptysis on postoperative day 17. A massive endobronchial hemorrhage during surveillance bronchoscopy occurred 39 days after transplantation. Immediate intervention, including rigid bronchoscopy followed by surgery, was effective in saving the patient. The pathophysiologic hypothesis to explain the fistula of the right pulmonary artery to the right mainstem bronchus probably involves ischemia of the anastomosis with necrosis of the suture zone complicated by endobronchial infection with Aspergillus. Rigid bronchoscopic intervention associated with an excellent medical surgical collaboration was pivotal in successfully rescuing the patient.
Key Words: bronchial aspergillosis • cystic fibrosis • massive hemoptysis • surgical treatment
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Introduction
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Truly massive hemoptysis fortunately is rare but, most often, is fatal. At this time, the therapeutic alternatives are limited. We report a case of massive hemoptysis following intrabronchial rupture of the right pulmonary artery 6 weeks after the patient underwent bilateral single-lung transplantation. The young patient was saved due to an effective collaboration between the endoscopy and surgery services.
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Case Report
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The patient was a 15-year-old girl who presented with terminal respiratory insufficiency secondary to cystic fibrosis. She was otherwise stable with no other significant active medical and surgical problems. On August 20, 1999, the patient underwent bilateral lung transplantation. The time of total ischemia of the graft was 7 h and 30 min. Medical treatment involved use of cyclosporine, azathioprine, and methylprednisolone, combined with empirical anti-infectious agents (IV ganciclovir and amphotericin B). The immediate postoperative period was significant for septicemia due to Alcaligenes xylosoxidans, which resolved with therapy with ceftazidime, tobramycin, and ciprofloxacin. Endobronchial Aspergillus colonization, for which therapy with inhaled amphotericin B was added, occurred mainly on the right bronchial suture and was found on the ninth day after the transplantation. The patient was weaned from mechanical ventilation and extubated on day 13.
On day 17, an episode of self-limited spontaneous hemoptysis was noted. Endoscopy revealed organized blood clots at the level of the right bronchial anastomosis and diffusely in the right peripheral bronchial tree (Fig 1
), as well as a whitish debris of Aspergillus at the level of the main bronchi. The episode of hemoptysis was attributed to endobronchial aspergillosis. A CT scan was performed, but the fistula was not seen. Antifungal treatment then was modified with increased doses of inhaled amphotericin B and the addition of oral itraconazole therapy.
On day 39, a planned rigid surveillance bronchoscopy with general anesthesia was performed. During a BAL, a massive endobronchial hemorrhage occurred with a total blood loss exceeding 1,500 mL from the level of the right bronchial anastomosis. The patient was placed immediately in the right lateral decubitus position to protect the uninvolved left lung and was suctioned continuously. The rigid endoscope was placed in the left mainstem bronchus to prevent contralateral lung compromise. However, hemorrhagic shock followed fluid resuscitation; probably the patient’s young age contributed to her survival on transfer to the operating room just 15 min after the initial bleeding occurred. An emergency repeat, clamshell, anterior thoracotomy was performed, and the right pulmonary artery was clamped between the ascending aorta and the superior vena cava. The clamping of the right main pulmonary artery resulted in the immediate improvement of oxygen saturation to 95%. An exploration of the anastomosis region revealed a right main pulmonary artery erosion with fistula to the right mainstem bronchus involving 5-cm segments of both structures. The eroded pulmonary artery was repaired and separated from the mainstem bronchus using a pedicled pericardial patch. Multiple transfusions to re-expand the volume resulted in favorable immediate postoperative recovery. Recurrent episodes of atelectasis of the right lower lobe caused by persistent blood clots were relieved by repeat bronchoscopy.
The endobronchial aspect gradually healed over a 4-month period. There was no recurrence of hemoptysis.
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Discussion
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There are many definitions of massive hemoptysis; however, most authors refer to asphyxia rather than to the hemodynamic or combined effect of both hemoptysis or asphyxia. Regardless, massive hemoptysis carries a very poor prognosis with a mortality rate exceeding 70% in the first 4 h.1
Massive hemoptysis after lung transplantation is rare, and we found only three cases in the literature.2
3
4
One case represented an arteriobronchial fistula.4
In the management of massive hemoptysis, maintenance of airway patency is of utmost importance. The patient must be placed immediately on the involved side to prevent contralateral lung compromise. The blood has to be evacuated constantly and efficiently to prevent asphyxia, which is most often the factor contributing to death by alveolar flooding1
and the formation of endobronchial blood clots. Hemorrhagic shock complicated by malignant arrhythmias, usually bradycardia, and ventricular fibrillation contributes to almost 100% mortality when occurring during the hemorrhagic event, partly because resuscitative techniques such as chest compressions and defibrillation can unfortunately render the local control of bleeding even more difficult.
The authors believe that rigid bronchoscopic intervention may be the only means to potentially improve outcome. Rigid bronchoscopic suction, tamponade using large-bore instruments, and the ability to ventilate simultaneously may be curative, as may bridge bronchoscopy with thoracic surgery. This is impossible with other methods. Such rigid bronchoscopy, however, must be practiced in a highly specialized environment with readily available dedicated anesthesiologists who are trained in endoscopic airway management and with a nearby thoracic surgery center in case a major problem (such as massive hemoptysis) arises. An immediate surgical intervention may be the only remaining alternative when a large-caliber vessel is involved.1
In the case presented, the ischemic anastomotic site became necrotic and subsequently colonized with Aspergillus. This could predispose the patient to fistula formation. The presentation of suture aspergillosis was first described by Kramer et al.5
In patients with cystic fibrosis, Aspergillus colonization is particularly common and may lead to the invasion of the anastomotic site.5
Whitish debris covering the suture line that is found on bronchoscopy is highly suggestive of this diagnosis. Such findings warrant sampling for Aspergillus and other mycoses. Systematic surveillance bronchoscopy in the postoperative period may be indicated to identify potential local complications of lung transplantation.6
Received for publication September 17, 2000.
Accepted for publication September 9, 2001.
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References
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Thompson, AB, Teschler, H, Rennard, SI (1992) Pathogenesis, evaluation and therapy for massive hemoptysis. Clin Chest Med 13,69-82[ISI][Medline]
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Hoff, SJ, Johnson, JE, Frist, WH (1993) Aortobronchial fistula after unilateral lung transplantation. Ann Thorac Surg 56,1402-1403[Abstract]
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Haydock, DA, Trulock, EP, Kaiser, LR, et al (1992) Lung transplantation: analysis of thirty-six consecutive procedures performed over a twelve-month period; the Washington University Lung Transplant Group. Thorac Cardiovasc Surg 103,329-340
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Kessler, R, Massard, G, Warter, A, et al (1997) Bronchial-pulmonary artery fistula after unilateral lung transplantation: a case report. J Heart Lung Transplant 16,674-677[ISI][Medline]
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Kramer, MR, Dennings, DW, Marshall, SE, et al (1991) Ulcerative tracheobronchiochitis after lung transplantation: a new form of invasive aspergillosis. Am Rev Respir Dis 144,552-556[ISI][Medline]
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Nunley, DR, Ohori, P, Grgurich, WF, et al (1998) Pulmonary aspergillosis in cystic fibrosis lung transplant recipients. Chest 114,1321-1329[Abstract/Free Full Text]
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Abstract
Introduction
