Eur J Cardiothorac Surg 2003;24:694-698
© 2003 Elsevier Science NL
The role of surgery in hemoptysis caused by thoracic actinomycosis; a forgotten disease
Ming-Shian Lu,
Hui-Ping Liu,
Chi-Hsiao Yeh,
Yi-Cheng Wu*,
Yun-Hen Liu,
Ming-Ju Hsieh,
Tzu-Ping Chen
Division of Thoracic and Cardiovascular Surgery, Chang Gung Memorial Hospital, 199 Tun-Hwa North Road, Taipei 105, Taiwan
Received 2 June 2003;
received in revised form 1 August 2003;
accepted 6 August 2003.
* Corresponding author. Tel.: +886-3-328-1200; fax: +886-3-328-5818
e-mail: expert96{at}ms11.hinet.net
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Abstract
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Objective: Thoracic actinomycosis is a relatively uncommon anaerobic infection caused by Actinomyces israelii. Rarely, it may be complicated by an unusual but significant hemoptysis manifestation. We describe our experiences of surgical treatment for hemoptysis in patients with pulmonary actinomycosis. Methods: From 1984 to 2002, 14 patients with thoracic actinomycosis and hemoptysis were diagnosed after thoracotomy. There were 11 male and three female patients with a mean age of 53.6 years. They all had non-specific symptoms and radiographic findings, and no case was an accurate diagnosis made at the time of hospital admission, including echo-guide aspiration, serial bronchoscopic biopsy and computed tomography biopsy. The indications of surgical intervention in our patients were for treating complications such as hemoptysis, chronic sinus discharge, or empyema, also limited to diagnostic purposes. Preoperative embolization was performed in seven cases; however, none was successful. Results: Among the 14 patients, all the lesions were unilateral. The disease was confined to the lung in all patients; and chest wall involvement in one patient. The surgical procedures included were as follows: pneumonectomy (n=1), bilobectomy (n=1), lobectomy (n=10), wedge resection (n=2), and lung lesion with concomitant chest wall resection and chest wall reconstruction (n=1). Mean intraoperative and postoperative blood loss within the first 24 h was 192.3 ml, and five patients required blood transfusion. All of the specimens from the lesion site showed histological evidence of Actinomyces infection. All patients regularly took 12 million units per day of intravenous crystalline penicillin G during their hospitalization, and oral procaine penicillin for up to 2 months. During the follow-up, all patients had an uneventful postoperative course. Conclusions: A high index of suspicion for this disease is advised for the surgeon engaged in the treatment of hemoptysis. Surgical resection of pulmonary actinomycosis is effective in preventing recurrence of hemoptysis. After diagnosis, it is treated using penicillin chemotherapy for at least 2 months.
Key Words: Thoracic actinomycosis • Hemoptysis • Sulfur granule
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1. Introduction
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Thoracic actinomycosis is a relatively uncommon anaerobic infection caused by Actinomyces israelii, which forms part of the normal flora in the oral cavity and intestine. This protean disease may present as chronic debilitating illness with radiographic manifestation simulating lung tumor, pulmonary infiltrating lesion, or chronic suppuration. Rarely, pulmonary actinomycosis may be complicated by an unusual but significant hemoptysis manifestation. Hemoptysis is a potentially lethal condition that must to be examined thoroughly and managed promptly. We describe herein our experiences of surgical treatment for pulmonary actinomycotic hemoptysis, with specific attention to the analysis of preoperative diagnosis.
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2. Patients and methods
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From 1984 to 2002, 14 patients with thoracic actinomycosis and hemoptysis were diagnosed after thoracotomy in Chang Gung Memorial Hospital. Charts were reviewed for clinical presentation, underlying lung disease, operative procedure, postoperative mortality, complications, and long-term outcome. There were 11 male and three female patients with a mean age of 53.6 years (ranging from 8 to 74 years). Chest pain, chronic cough, and fever were also common complaints by patients. Despite series examinations for these patients, including echo-guide aspiration, serial bronchoscopic biopsy and computed tomography (CT) biopsy, no accurate diagnosis could be made at the time of hospital admission. Final diagnosis was based on histological examination of the resected tissue after surgery.
Preoperative chest radiographs were categorized as mass (n=7), pulmonary infiltration or consolidation (n=5), abscess formation (n=1), and chest wall lesion (n=1). The surgical approach was decided on because of the following symptoms, All patients had hemoptysis with cough and bronchitis in association, and seven (50%) complained of chronic cough only or associated with low-grade fever. Eleven of the 14 patients presenting hemoptysis had a history of minor and recurrent hemoptysis for weeks or months before operation, whereas the remaining three patients had a single episode of bleeding. Seven hemoptysis cases were severe, demonstrating bleeding exceeding 200 ml. Preoperative embolization was performed in seven cases; however, no case was successful. Despite the surgical indication for hemoptysis, these patients also presented with a clinical picture of suspected primary lung tumor initially. Therefore surgical intervention provided the tissue proof simultaneously. The clinical diagnosis of actinomycosis was not suspected in any of the 14 cases, and actinomycosis was diagnosed by the histologic study of the surgically obtained specimen.
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3. Results
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Among the 14 patients, all of the lesions were unilateral. The disease was confined to the lung in 14; the chest wall associated with the pleura was involved in one patient. The surgical intervention procedures included pneumonectomy (n=1), bilobectomy (n=1), lobectomy (n=10), wedge resection (n=2), and one lung lesion with concomitant chest wall resection and chest wall reconstruction (Table 1). Mean intraoperative and postoperative blood loss within the first 24 h was 192.3 ml (range 100–600 ml), and five patients (35.7%) required blood transfusion. All of the specimens from the lesion site showed histological evidence of Actinomyces infection with the picture of ‘sulfur granules’ and ‘branching filaments’. All patients regularly took 12 million units per day of intravenous crystalline penicillin G during their hospitalization. After that, antibiotic treatment was completed in up to 3 months with oral procaine penicillin. During the follow-up, all patients had an uneventful postoperative course without any evidence of recurrence.
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4. Discussion
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Pulmonary actinomycosis is a disease that is difficult to diagnose with its radiographic manifestation simulating lung tumor (Fig. 1) , pulmonary infiltrating lesion, or chronic suppurated appearance [1]. Clinically, it may simulate a wide variety of pulmonary disorders. Sputum culture and bronchoalveolar lavage are inadequate for the diagnosis of pulmonary actinomycosis because Actinomyces may form part of the normal flora [2], and it has been reported that the presence of physiologic saline solution, commonly used in bronchoalveolar lavage, inhibits the growth of pathogenic Actinomyces [3]. The reported accuracy of the biopsy methods, transbronchial and needle aspiration, are low owing to inadequate aspiration [1]. In our present study, the patients encountered the same diagnostic problems. The diagnosis of thoracic actinomycosis involving the lung is usually hampered by difficulty in isolating the organism. It has been shown that isolation from the bronchial brush or sputum studies are difficult; even through careful biopsy. The specimen may not exactly contain the areas of ‘sulfur granules’ or ‘branching filaments’. Therefore the majority of patients are diagnosed postsurgically by pathological proof. However, we suggest that two or more aspirations or biopsies are needed to obtain accurate bacteriologic or histologic diagnosis so as to prevent unnecessary surgery.
Thoracic actinomycosis is uncommon to most practicing thoracic surgeons. It is a rare disease caused by Actinomyces israellii, characterized by suppuration, sinus tract formation, and purulent discharge containing yellowish ‘sulfur granules’ (Fig. 2)
; however, sulfur granules are not unique to actinomycosis. The absence of sulfur granules from any lesion does not exclude the diagnosis of actinomycosis. Sulfur granules have been reported absent in a proven case of cerebral actinomycosis [4]. Thoracic actinomycosis is believed to invade the thoracic organ through the oropharynx causing bronchopulmonary infection as well as pleural and chest wall involvement [5,6]. The clinical symptoms of thoracic actinomycosis frequently include fever, chills, hemoptysis, cough, pleuritic chest pain, and anemia. It may present as empyema thoracis, empyema necessitates, chronic draining sinus tract with typical ‘sulfur granules’ contents (Fig. 3)
, superior vena cava syndrome, or pericardial effusion [7,8] in advanced disease.
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Fig. 2. Histopathology examination of lung demonstrating aggregates of ‘sulfur granule’ (H&E stain, x400).
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Fig. 3. Chest CT demonstrating a consolidation of anterior segment of right upper lobe contiguous to pleura and chest wall. Prominent chest wall soft tissue with low attenuation change.
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Chronic inflammatory lung diseases and bronchogenic carcinoma remain the most common causes of hemoptysis in the United States [9]. Massive hemoptysis most commonly occurs from bronchogenic carcinoma, tuberculosis, lung abscess and aspergilloma [10]. However, there has been a limited number of reports focusing upon this protean disease. When the tracheobronchial tree is flooded by severe hemoptysis, life-threatening asphyxiation may occur subsequently and emergency intensive care unit admission for the patient must be arranged. A massive hemoptysis warrants prompt medical intervention because of reduced alveolar gas exchange and the unstable hemodynamic condition that may ensue. It is estimated that 400 ml of blood in the alveolar space is sufficient to cause significant hindrance to the oxygen transfer [11], although only minor changes in the vital signs will be noted for the same amount of blood loss [12]. Prior to definitive therapy, however, the risk remains in spite of cessation of an episode of massive hemoptysis owing to its unpredictable rate of relapse.
Bronchial artery embolization (BAE), first performed by Remy et al. in 1973 [13], is now considered the most effective non-surgical treatment in massive hemoptysis because of immediate and long-term results, and since then the surgical treatment in the management of these patients has been reduced. There are various other options to control the hemorrhage, including bronchoscopic cold saline solution lavage [14], endobronchial balloon tamponade with or without endobronchial instillation of epinephrine [15,16], instillation of thrombin or fibrinogen-thrombin infusions endobronchially [17], radiotherapy [18], and laser photocoagulation [19]. Nevertheless, surgery still plays the critical role in the treatment of massive hemoptysis caused by many kinds of etiologies [20,21]. Knott-Craig et al. [22] have shown that 36.4% of patients suffered from recurrent hemoptysis after medical therapy within 6 months of hospital discharge. Almost one half of these recurrent episodes proved fatal. Hence surgical resection still remains the curative option. Before surgical intervention, the benefits should always be weighed against the risk of complications and morbidity in all potential surgical candidates, especially those with significant underlying systemic diseases.
There are three vascular networks that may cause blood flooding into the tracheobronchial tree: the bronchial, the non-bronchial collateral systemic vessels, and the pulmonary arterial system. An inflammatory pulmonary disease quite often induces hemoptysis originating from neovascularization of the bronchial arterial system. It has been reported that the bronchial circulation, rather than the pulmonary arterial system, is the usual source of bleeding from areas of acute and chronic lung inflammation and scarring. This vascular system can increase to more than one third of the systemic flow in chronic pulmonary disease [23]. Keller et al. [24] reported that 45% of 20 patients with massive or recurrent hemoptysis had a significant blood supply from non-bronchial collateral vessels. Hemoptysis from the pulmonary arterial system comes through an ulceration of the vascular wall caused by pulmonary destructive processes whatever the pathogenesis could be. Due to the endowed mural musculature of the neovascularization, bleeding from the bronchial and the non-bronchial collateral systemic vessels could be managed by pharmacological methods (vasoactive drugs such as vasopressin or aerosolized adrenaline) or by physical methods (bronchial cold saline lavage) to produce vessel contraction and subsequent temporary slowing or cessation of the bleeding. However, the pulmonary arterial vessels may not be able to vasospasm as effectively as bronchial ones. Hence the pharmacological or physical way may not be appropriate for this kind of origin, and an operation must be performed emergently after careful evaluation of the risk of complications. The arterial source of hemoptysis in patients with actinomycosis may be a mixture of the three vascular networks. It is difficult to predict the origin of hemoptysis and decide the appropriate treatment policy. The indications for surgical intervention in our patients were for treating complications such as hemoptysis, chronic sinus discharge, or empyema, also limited to diagnostic purposes. Seven out of the 14 patients presented radiographic evidence of lung mass, where malignancy could not be excluded, and besides, actinomycosis might coexist with lung cancer [25]; therefore obtaining tissue for pathologic examination is imperative.
A high index of suspicion for this disease is advised for the thoracic surgeon engaged in the treatment of hemoptysis. The clinical approach for the management of massive hemoptysis should be guided by the underlying etiology. Actinomyces israelii infections should be suspected in chronic infiltrative, nodular, cavitary process and tumor-like lesions, in addition to other most probable causes. Surgical intervention is indicated in those with recurrent hemoptysis or uncertain diagnosis of cancer. Resection of pulmonary actinomycosis is effective in preventing recurrence of hemoptysis. We suggest that surgical treatment with appropriate penicillin chemotherapy may offer a good chance of cure with low morbidity and mortality.
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Abstract
1. Introduction
