- Email: [email protected]
Endobronchial Actinomycosis Associated With Foreign Body
19 Brinson GM, Noone PG, Mauro MA, et al. Bronchial artery embolization for the treatment of hemoptysis in patients with cystic fibrosis. Am J Respir Crit Care Med 1998; 157:1951– 1958 20 Roberts AC. Bronchial artery embolization therapy. J Thorac Imaging 1990; 5:60 –72 21 Tanaka N, Yamakado K, Murashima S, et al. Superselective bronchial artery embolization for hemoptysis with a coaxial microcatheter system. J Vasc Interv Radiol 1997; 8:65–70 22 Shneerson JM, Emerson PA, Phillips RH. Radiotherapy for massive hemoptysis from an aspergilloma. Thorax 1980; 35:953–954 23 Yeoh CB, Hubaytar RT, Ford JM. Treatment of massive hemoptysis in pulmonary tuberculosis. J Thorac Cardiovasc Surg 1967; 54:503–510 24 Sehhat S, Oreizie M, Moinedine K. Massive pulmonary hemorrhage: surgical approach as a choice of treatment. Ann Thorac Surg 1978; 25:12–15 25 Knott-Craig CJ, Oostuizen G, Rossouw G, et al. Management and prognosis of massive hemoptysis: recent experience with 120 patients. J Thorac Cardiovasc Surg 1993; 105:394 –397 26 Giray CB, Us D, Gunay C, et al. Antibacterial and cytotoxic effects of n-butyl-2-cyanoacrylate used as a tissue adhesive [Turkish]. Mikrobiyol Bul 1993; 27:154 –163 27 Eastman DP, Robicsek F. Application of cyanoacrylate adhesive (Krazy Glue) in critical cardiac injuries. J Heart Valve Dis 1998; 7:72–74 28 Sung JJ, Yeo W, Suen R, et al. Injection sclerotherapy for variceal bleeding in patients with hepatocellular carcinoma: cyanoacrylate versus sodium tetradecyl sulphate. Gastrointest Endosc 1998; 47:235–239 29 Eng J, Sabanathan S. Tissue adhesive in bronchial closure. Ann Thorac Surg 1989; 48:683– 685 30 Papathiofenis FJ. Prothrombotic cytotoxicity of cyanoacrylate tissue adhesive. J Surg Res 1989; 47:309 –312 31 Kopferschmit-Kuber MC, Stenger R, Blaumeiser M, et al. Asthma, rhinitis, and urticaria following occupational exposure to cyanoacrylate glues. Rev Mal Respir 1996; 13:305–307 32 Conde-Salazar L, Rojo S, Guimaraens D. Occupational allergic contact dermatitis from cyanoacrylate. Am J Contact Dermat 1998; 9:188 –189
Endobronchial Actinomycosis Associated With Foreign Body* Four Cases and a Review of the Literature Ste´phane Chouabe; Dominique Perdu, MD; Gae´tan Desle´e, MD; Dragisa Milosevic, MD; Elisabeth Marque, MD; and Franc¸ois Lebargy, MD, PhD
Four cases of primary endobronchial actinomycosis associated with an inhaled foreign body are described. In the light of these cases and those previously reported in the literature, we describe the main features of this uncommon association. All patients were > 55 years old, were predominantly men, and were usually in a debilitated state. In > 50% of cases, the clinical presentation was suggestive of lung cancer. Thoracic CT rarely revealed a www.chestjournal.org
foreign body, but the granulomatous reaction of the bronchial wall was sometimes suggestive of bronchial thickening. Sulfur granules identified on bronchial biopsies were highly suggestive of actinomycosis in most cases, but microbiological culture findings were usually negative. Antibiotics generally ensure good recovery. Extraction of the foreign body was delayed after antibiotic therapy in one half of cases, suggesting the need for endoscopic follow-up in bronchial actinomycosis. (CHEST 2002; 121:2069 –2072) Key words: actinomycosis; bronchus; endoscopy; foreign body; thoracic CT
is a chronic suppurative infection due to A ctinomycosis a group of anaerobic or microaerophilic bacteria be-
longing to the resident flora of the oropharynx and GI tract. Actinomyces israelii is the main representative pathogen in human infection, although most cases of actinomycosis are thought to be polymicrobial. Twenty percent of cases of actinomycosis are located in the thorax.1 Primary endobronchial actinomycosis is rare and very uncommon in association with foreign body aspiration.2 In this context, bronchial infection is thought to result from direct aspiration of an Actinomyces-contaminated foreign body. We report four cases of endobronchial actinomycosis associated with bronchial foreign body. In the light of these cases and those previously reported in the literature, we describe the diagnostic, pathologic, bacteriological and therapeutic features of this association, which must be considered more systematically.
Case Reports Between January 1996 to September 1999, four cases of endobronchial actinomycosis associated with foreign body were observed at Reims University Hospital. Clinical data, diagnostic procedures, and treatments are summarized in Table 1. These four patients were men between the ages of 56 and 79 years (mean age, 70 years) with a history of smoking (19 to 33 pack-years). Each patient presented in a debilitated state: metastatic cervical node (patient 1), noninsulin-dependent diabetes mellitus (patient 2), previous lung cancer treated by upper and middle lobectomy 1 year previously (patient 3), and alcoholism (patient 4). Presenting symptoms were left chest pain with fever related to pleural effusion (patient 2) and persistent purulent bronchitis (patients 3 and 4). One patient (patient 1) had no respiratory symptoms. None of the patients reported any history of choking. Laboratory data revealed a raised leukocyte count (11.9 ⫻ 106/L) in one case (patient 2) and inflammatory signs in two cases (patient 2 and patient 4). The pleural effusion in patient 2 was a cloudy exudate containing 1,500 leukocytes per microliter with a predominance of polymorphonuclear leukocytes. Chest radiography revealed a tooth in the right hilar region in *From the Department of Respiratory and Allergic Diseases, Hoˆ pital de la Maison Blanche, 51092 Reims, France. Manuscript received January 11, 2001; revision accepted January 22, 2002. Correspondence to: Franc¸ois Lebargy, MD, PhD, Service de Pneumologie, CHU de la Maison Blanche, 45 rue Cognacq Jay, 51092 Reims, France; e-mail: [email protected] CHEST / 121 / 6 / JUNE, 2002
2069
2070
Selected Reports
1 57
Female ND ND ND
Hemoptysis, dyspnea
Bronchial opacity
Mass obstructing the lower right bronchus
Fish bone
Cases, No. Age, yr Sex Smoking Alcohol Associated disease
Symptoms
Chest radiographs
Bronchoscopy
Foreign body
*ND ⫽ not determined.
Umeki et al3
Features
Chicken bone
Mass obstructing the left upper bronchus
Left upper bronchial opacity
Cough, hemoptysis, dyspnea
Male No ND Diabetes mellitus, poor dental hygiene
1 56
Mignon et al9
Chicken bone
Yellowish obstructive mass
Male Yes ND Diabetes mellitus, mental retardation, poor dental hygiene Cough, recurrent pneumonia, hemoptysis Right base opacity, bronchiectasis
1 58
Julia et al7
Chicken bone
Right base opacity, mediastinal adenopathy, atelectasis Mass obstructing the right main bronchus
Cough, recurrent pneumonia
Male ND No Diabetes mellitus, poor dental hygiene
1 66
Dicpinigaitis et al8
Mass obstructing the lower right bronchus
Chicken bone
Bone fragment (n ⫽ 1), dental crown (n ⫽ 1)
Cough, recurrent pneumonia, hemoptysis Right base opacity, mediastinal opacity
Male ND ND Epilepsy
1 ND
Mingrone et al5
Mass obstructing the right main bronchus (n ⫽ 2)
Cough (n ⫽ 2), fever (n ⫽ 1), purulent sputum (n ⫽ 1) Right atelectasis (n ⫽ 1), right pleural effusion (n ⫽ 2)
2 57, 65 Female (n ⫽ 1)/male (n ⫽ 1) Yes (n ⫽ 1)/no (n ⫽ 1) No (n ⫽ 1) Allergy to tetracyclines (n ⫽ 1)
Fernandez et al6
Table 1—Bronchial Actinomycosis Associated With Foreign Body: Clinical, Radiologic, and Endoscopic Features*
Mass obstructing the right main bronchus (n ⫽ 1), lower left (n ⫽ 1) and lower right bronchus (n ⫽ 2) Tooth (n ⫽ 1), chicken bone (n ⫽ 1), grape seeds (n ⫽ 1), bean (n ⫽ 1)
Pain (n ⫽ 1), chronic bronchitis (n ⫽ 1), cough (n ⫽ 1) Bronchial opacity (n ⫽ 3), left atelectasis (n ⫽ 1), right atelectasis (n ⫽ 1), pleural effusion (n ⫽ 1)
4 56, 69, 78, 79 Male (n ⫽ 4) Yes (n ⫽ 4) Yes (n ⫽ 2) Neoplasia (n ⫽ 2), poor dental hygiene (n ⫽ 1)
Current Study
Figure 2. Thoracic CT showing high-density endoluminal mass in the right main bronchus.
Discussion
Figure 1. Chest radiograph showing a foreign body (tooth) in the right hilar area (arrow).
one patient (patient 1) [Fig 1]. Related images, such as a left pleural effusion associated with lower lobe consolidation or an alveolar opacity in the right lower lobe, were the main presenting signs in two patients (patient 2 and patient 4) and were confirmed by CT. Chest radiographic findings were normal in the last patient (patient 3). Thoracic CT showed calcified material corresponding to a tooth in the bronchus intermedius in one case (patient 1). No foreign bodies were observed in the other cases, but an obstructive high-density mass was seen in the lumen of the left lower lobe (Fig 2) or right main bronchus (patient 2 and patient 4). In patient 3, with normal chest radiographic finding, CT showed segmental thickening of the right bronchus. Fiberoptic bronchoscopy revealed a mass suggestive of a tumor obstructing 40 to 60% of bronchial lumen in two cases (patient 1 and patient 2), but the foreign bodies (a tooth and a chicken bone) were not identified at the initial examination. They were only visualized after 3 to 4 months of antibiotic therapy, and were removed by cryotherapy in one case and biopsy forceps in the other case. In the two remaining cases (patient 3 and patient 4), the bronchial mucosa was inflamed and necrotic. The foreign body was identified and immediately removed (grape seeds and a bean). In all cases, bronchial biopsies revealed a granulomatous inflammatory reaction in the mucosa. Periodic acid-Schiff– positive filaments consistent with actinomycosis infection were identified on bronchial biopsies in two cases, and sulfur granules were detected in one case. In the last patient, periodic acidSchiff–positive filaments were identified only on cytologic examination of paraffin-embedded respiratory tract secretions. All patients were treated with antibiotics for 3 to 6 months: amoxicillin, 3 g/d, in three cases, and erythromycin in one case because of documented allergy to -lactam antibiotics. A favorable course was observed after prolonged antibiotic therapy and foreign body extraction in three cases, but the remaining patient died 3 months later from Pseudomonas aeruginosa pneumonia associated with chemotherapy-induced neutropenia. www.chestjournal.org
Bronchial involvement is a rare form of thoracic actinomycosis3 that has sometimes been reported to be associated with foreign bodies. The four cases presented here and the seven cases previously reported3,5–9 illustrate the main clinical features of this association (Table 1). All patients were ⬎ 55 years old and presented a male predominance (nine men and two women). This sex ratio has also been reported for thoracic actinomycosis.1 Two types of situations predisposing to actinomycosis are reported: debilitated state, such as noninsulin-dependent diabetes mellitus (27%); neoplasm (18%), or poor dental hygiene (36%); and conditions that facilitate foreign body aspiration, such as mental retardation (9%). The main symptoms are cough (63%), recurrent pneumonia (27%), and hemoptysis (36%). A history of choking was never reported in any case of foreign body aspiration associated with actinomycosis, but a large series of bronchial foreign body aspiration in adults reports choking in only 50% cases.10 Thoracic CT showed a radiopaque foreign body in two of our cases and in one previously reported case (two chicken bones and one tooth, respectively), using endoluminal three-dimensional reconstruction in one case.9 Bronchial involvement consisting of a thickened bronchial wall was identified in three of our cases, and has been previously reported in two cases in the literature.4,9 Other indirect signs are seen on CT scan: dense pulmonary alveolar opacity (45%), atelectasis (36%), pleural effusion (18%), or bronchiectasis (9%). Endoscopically, an obstructive endoluminal mass was found in all cases, and the foreign body was detected immediately in only 45% of cases. This type of presentation, suggestive of lung cancer, is frequently reported in endobronchial actinomycosis not associated with foreign body.11 It therefore seems important to perform follow-up bronchoscopy after antibiotic therapy to exclude the presence of a foreign body. Actinomycosis infection was diagnosed on bronchial biopsies in all cases. The presence of sulfur granules in biopsy samples is highly suggestive of actinomycosis.12 In CHEST / 121 / 6 / JUNE, 2002
2071
contrast, culture findings are usually negative, as A israelii is a strict anaerobe and is frequently associated with nonanaerobic contaminants. Bronchial aspiration samples may also be contaminated by oral flora. A israelii is usually sensitive to penicillins. Other effective antibiotic families are tetracyclines, lincosamides, and trimethoprim-sulfamethoxazole.12 The optimal duration of treatment has not been clearly established, but treatment for at least 45 days seems to be necessary in the case of thoracic actinomycosis.1 Our patients were treated with penicillin G or ampicillin, but one patient allergic to -lactam antibiotics was treated with a macrolide. Clindamycin can be an alternative therapy in these situations. Bronchoscopic removal of the foreign body was effective in all cases. However, in 45% of cases, the foreign body was only detected some time after starting antibiotics. Extraction procedures required bronchial aspiration (3 of 11 cases), biopsy forceps (6 of 11 cases), YAG laser (1 of 11 cases) or cryotherapy (1 of 11 cases). This review of the literature concerning endobronchial actinomycosis associated with foreign body aspiration confirms the development of this infection in debilitated patients with poor oral hygiene, frequently mimicking lung cancer. Endobronchial actinomycosis is usually diagnosed on histologic examination of bronchial biopsies and requires investigation of an associated bronchial foreign body, which may be missed on the initial assessment.
References 1 Hsieh MJ, Liu HP, Chang JP, et al. Thoracic actinomycosis. Chest 1993; 104:366 –370 2 Dalhoff K, Wallner S, Finck C, et al. Endobronchial actinomycosis. Eur Respir J 1994; 7:1189 –1191 3 McHardy G, Browne DC. Primary bronchial actinomycosis. South Med J 1943; 36:674 – 676 4 Umeki S, Nakajima M, Tsukiyama K, et al. Foreign bodyinduced bronchial actinomycosis with severe stenosis that must be distinguished from lung cancer. Nihon Kyobu Shikkan Gakkai Zasshi 1990; 28:481– 486 5 Mingrone H, Perrone R, La Rosa S, et al. Primary bronchial actinomycosis and foreign body. Medicina (B Aires) 1995; 55:337–340 6 Fernandez Jorge MA, Castro Villamor MA, Bartolome de Castro EM, et al. Thoracic actinomycosis and intrabronchial foreign body: presentation of two cases. An Med Interna 1995; 12:79 – 81 7 Julia G, Rodriguez de Castro F, Caminero J, et al. Endobronchial actinomycosis associated with a foreign body. Respiration 1991; 58:229 –230 8 Dicpinigaitis PV, Bleiweiss IJ, Krellenstein DJ, et al. Primary endobronchial actinomycosis in association with foreign body aspiration. Chest 1992; 101:283–285 9 Mignon F, Mesurolle B, Chambellan A, et al. Granulomatous reaction to a foreign body mimicking bronchogenic tumor: CT findings with virtual endoscopy. J Radiol 1997; 78:1181– 1184 10 Lan RS. Non-asphyxiating tracheobronchial foreign bodies in adults. Eur Respir J 1994; 7:510 –514 11 Ariel I, Breuer R, Kamal NS, et al. Endobronchial actinomycosis simulating bronchogenic carcinoma. Chest 1991; 99: 493– 495 12 Brown JR. Human actinomycosis: a study of 181 subjects. Hum Pathol 1973; 4:319 –330 2072
Progression of Idiopathic Pulmonary Fibrosis in Native Lungs After Single Lung Transplantation* Momen M. Wahidi, MD; James Ravenel, MD†; Scott M. Palmer, MD, MHS, FCCP; and H. Page McAdams, MD
This retrospective, single-center study was conducted to assess the response of native idiopathic pulmonary fibrosis (IPF) lungs to a potent cyclosporine-based immunosuppressive regimen in single-lung transplantation recipients. The study included IPF patients who had undergone single-lung transplantation and had chest CT scans before and after transplantation. Five patients underwent single-lung transplantation for IPF between April 1992 and January 2001, and met entry criteria. All patients were placed on an immunosuppressive regimen consisting of prednisone, azathioprine, and cyclosporine. In two of the five patients, ground glass attenuation in the native IPF lung improved post-transplantation. However, fibrotic changes progressed in all five patients. In patients with advanced IPF, a potent cyclosporine-based immunosuppressive regimen is not likely to have an effect on the progression of the disease. (CHEST 2002; 121:2072–2076) Key words: cyclosporine; idiopathic pulmonary fibrosis; lung transplantation Abbreviations: HRCT ⫽ high-resolution CT; IPF ⫽ idiopathic pulmonary fibrosis
pulmonary fibrosis (IPF) is an incurable I diopathic disease with a 5-year survival rate of 30 to 50% from the
time of diagnosis.1 To date, there are no sufficient data to support a favorable effect of any treatment on the progressive course of IPF. Corticosteroids have become the conventional treatment despite their limited efficacy.2 It is estimated that 15 to 30% of patients with IPF improve while receiving corticosteroids.3–5 Azathioprine has been used as a second-line therapeutic agent in IPF patients with poor responses or intolerance to corticosteroids. In one study, Raghu et al6 randomized patients to prednisone and azathioprine or to prednisone and placebo, and found a trend toward an improved survival rate in the combinedtreatment group. Several case reports7–10 have documented an encouraging response to cyclosporine in IPF patients. *From the Department of Internal Medicine (Drs. Wahidi and Palmer), Division of Pulmonary and Critical Care, and the Department of Radiology (Drs. Ravenel and McAdams), Duke University Medical Center, Durham, NC. †Now at the Medical University of South Carolina, Columbia, SC. Manuscript received September 11, 2001; revision accepted January 15, 2002. Correspondence to: Momen M. Wahidi, MD, Division of Pulmonary and Critical Care, Duke University Medical Center, Box 3221, Durham, NC 27710; e-mail: [email protected] Selected Reports
Endobronchial Actinomycosis Associated With Foreign Body* Four Cases and a Review of the Literature Ste´phane Chouabe; Dominique Perdu, MD; Gae´tan Desle´e, MD; Dragisa Milosevic, MD; Elisabeth Marque, MD; and Franc¸ois Lebargy, MD, PhD
Four cases of primary endobronchial actinomycosis associated with an inhaled foreign body are described. In the light of these cases and those previously reported in the literature, we describe the main features of this uncommon association. All patients were > 55 years old, were predominantly men, and were usually in a debilitated state. In > 50% of cases, the clinical presentation was suggestive of lung cancer. Thoracic CT rarely revealed a www.chestjournal.org
foreign body, but the granulomatous reaction of the bronchial wall was sometimes suggestive of bronchial thickening. Sulfur granules identified on bronchial biopsies were highly suggestive of actinomycosis in most cases, but microbiological culture findings were usually negative. Antibiotics generally ensure good recovery. Extraction of the foreign body was delayed after antibiotic therapy in one half of cases, suggesting the need for endoscopic follow-up in bronchial actinomycosis. (CHEST 2002; 121:2069 –2072) Key words: actinomycosis; bronchus; endoscopy; foreign body; thoracic CT
is a chronic suppurative infection due to A ctinomycosis a group of anaerobic or microaerophilic bacteria be-
longing to the resident flora of the oropharynx and GI tract. Actinomyces israelii is the main representative pathogen in human infection, although most cases of actinomycosis are thought to be polymicrobial. Twenty percent of cases of actinomycosis are located in the thorax.1 Primary endobronchial actinomycosis is rare and very uncommon in association with foreign body aspiration.2 In this context, bronchial infection is thought to result from direct aspiration of an Actinomyces-contaminated foreign body. We report four cases of endobronchial actinomycosis associated with bronchial foreign body. In the light of these cases and those previously reported in the literature, we describe the diagnostic, pathologic, bacteriological and therapeutic features of this association, which must be considered more systematically.
Case Reports Between January 1996 to September 1999, four cases of endobronchial actinomycosis associated with foreign body were observed at Reims University Hospital. Clinical data, diagnostic procedures, and treatments are summarized in Table 1. These four patients were men between the ages of 56 and 79 years (mean age, 70 years) with a history of smoking (19 to 33 pack-years). Each patient presented in a debilitated state: metastatic cervical node (patient 1), noninsulin-dependent diabetes mellitus (patient 2), previous lung cancer treated by upper and middle lobectomy 1 year previously (patient 3), and alcoholism (patient 4). Presenting symptoms were left chest pain with fever related to pleural effusion (patient 2) and persistent purulent bronchitis (patients 3 and 4). One patient (patient 1) had no respiratory symptoms. None of the patients reported any history of choking. Laboratory data revealed a raised leukocyte count (11.9 ⫻ 106/L) in one case (patient 2) and inflammatory signs in two cases (patient 2 and patient 4). The pleural effusion in patient 2 was a cloudy exudate containing 1,500 leukocytes per microliter with a predominance of polymorphonuclear leukocytes. Chest radiography revealed a tooth in the right hilar region in *From the Department of Respiratory and Allergic Diseases, Hoˆ pital de la Maison Blanche, 51092 Reims, France. Manuscript received January 11, 2001; revision accepted January 22, 2002. Correspondence to: Franc¸ois Lebargy, MD, PhD, Service de Pneumologie, CHU de la Maison Blanche, 45 rue Cognacq Jay, 51092 Reims, France; e-mail: [email protected] CHEST / 121 / 6 / JUNE, 2002
2069
2070
Selected Reports
1 57
Female ND ND ND
Hemoptysis, dyspnea
Bronchial opacity
Mass obstructing the lower right bronchus
Fish bone
Cases, No. Age, yr Sex Smoking Alcohol Associated disease
Symptoms
Chest radiographs
Bronchoscopy
Foreign body
*ND ⫽ not determined.
Umeki et al3
Features
Chicken bone
Mass obstructing the left upper bronchus
Left upper bronchial opacity
Cough, hemoptysis, dyspnea
Male No ND Diabetes mellitus, poor dental hygiene
1 56
Mignon et al9
Chicken bone
Yellowish obstructive mass
Male Yes ND Diabetes mellitus, mental retardation, poor dental hygiene Cough, recurrent pneumonia, hemoptysis Right base opacity, bronchiectasis
1 58
Julia et al7
Chicken bone
Right base opacity, mediastinal adenopathy, atelectasis Mass obstructing the right main bronchus
Cough, recurrent pneumonia
Male ND No Diabetes mellitus, poor dental hygiene
1 66
Dicpinigaitis et al8
Mass obstructing the lower right bronchus
Chicken bone
Bone fragment (n ⫽ 1), dental crown (n ⫽ 1)
Cough, recurrent pneumonia, hemoptysis Right base opacity, mediastinal opacity
Male ND ND Epilepsy
1 ND
Mingrone et al5
Mass obstructing the right main bronchus (n ⫽ 2)
Cough (n ⫽ 2), fever (n ⫽ 1), purulent sputum (n ⫽ 1) Right atelectasis (n ⫽ 1), right pleural effusion (n ⫽ 2)
2 57, 65 Female (n ⫽ 1)/male (n ⫽ 1) Yes (n ⫽ 1)/no (n ⫽ 1) No (n ⫽ 1) Allergy to tetracyclines (n ⫽ 1)
Fernandez et al6
Table 1—Bronchial Actinomycosis Associated With Foreign Body: Clinical, Radiologic, and Endoscopic Features*
Mass obstructing the right main bronchus (n ⫽ 1), lower left (n ⫽ 1) and lower right bronchus (n ⫽ 2) Tooth (n ⫽ 1), chicken bone (n ⫽ 1), grape seeds (n ⫽ 1), bean (n ⫽ 1)
Pain (n ⫽ 1), chronic bronchitis (n ⫽ 1), cough (n ⫽ 1) Bronchial opacity (n ⫽ 3), left atelectasis (n ⫽ 1), right atelectasis (n ⫽ 1), pleural effusion (n ⫽ 1)
4 56, 69, 78, 79 Male (n ⫽ 4) Yes (n ⫽ 4) Yes (n ⫽ 2) Neoplasia (n ⫽ 2), poor dental hygiene (n ⫽ 1)
Current Study
Figure 2. Thoracic CT showing high-density endoluminal mass in the right main bronchus.
Discussion
Figure 1. Chest radiograph showing a foreign body (tooth) in the right hilar area (arrow).
one patient (patient 1) [Fig 1]. Related images, such as a left pleural effusion associated with lower lobe consolidation or an alveolar opacity in the right lower lobe, were the main presenting signs in two patients (patient 2 and patient 4) and were confirmed by CT. Chest radiographic findings were normal in the last patient (patient 3). Thoracic CT showed calcified material corresponding to a tooth in the bronchus intermedius in one case (patient 1). No foreign bodies were observed in the other cases, but an obstructive high-density mass was seen in the lumen of the left lower lobe (Fig 2) or right main bronchus (patient 2 and patient 4). In patient 3, with normal chest radiographic finding, CT showed segmental thickening of the right bronchus. Fiberoptic bronchoscopy revealed a mass suggestive of a tumor obstructing 40 to 60% of bronchial lumen in two cases (patient 1 and patient 2), but the foreign bodies (a tooth and a chicken bone) were not identified at the initial examination. They were only visualized after 3 to 4 months of antibiotic therapy, and were removed by cryotherapy in one case and biopsy forceps in the other case. In the two remaining cases (patient 3 and patient 4), the bronchial mucosa was inflamed and necrotic. The foreign body was identified and immediately removed (grape seeds and a bean). In all cases, bronchial biopsies revealed a granulomatous inflammatory reaction in the mucosa. Periodic acid-Schiff– positive filaments consistent with actinomycosis infection were identified on bronchial biopsies in two cases, and sulfur granules were detected in one case. In the last patient, periodic acidSchiff–positive filaments were identified only on cytologic examination of paraffin-embedded respiratory tract secretions. All patients were treated with antibiotics for 3 to 6 months: amoxicillin, 3 g/d, in three cases, and erythromycin in one case because of documented allergy to -lactam antibiotics. A favorable course was observed after prolonged antibiotic therapy and foreign body extraction in three cases, but the remaining patient died 3 months later from Pseudomonas aeruginosa pneumonia associated with chemotherapy-induced neutropenia. www.chestjournal.org
Bronchial involvement is a rare form of thoracic actinomycosis3 that has sometimes been reported to be associated with foreign bodies. The four cases presented here and the seven cases previously reported3,5–9 illustrate the main clinical features of this association (Table 1). All patients were ⬎ 55 years old and presented a male predominance (nine men and two women). This sex ratio has also been reported for thoracic actinomycosis.1 Two types of situations predisposing to actinomycosis are reported: debilitated state, such as noninsulin-dependent diabetes mellitus (27%); neoplasm (18%), or poor dental hygiene (36%); and conditions that facilitate foreign body aspiration, such as mental retardation (9%). The main symptoms are cough (63%), recurrent pneumonia (27%), and hemoptysis (36%). A history of choking was never reported in any case of foreign body aspiration associated with actinomycosis, but a large series of bronchial foreign body aspiration in adults reports choking in only 50% cases.10 Thoracic CT showed a radiopaque foreign body in two of our cases and in one previously reported case (two chicken bones and one tooth, respectively), using endoluminal three-dimensional reconstruction in one case.9 Bronchial involvement consisting of a thickened bronchial wall was identified in three of our cases, and has been previously reported in two cases in the literature.4,9 Other indirect signs are seen on CT scan: dense pulmonary alveolar opacity (45%), atelectasis (36%), pleural effusion (18%), or bronchiectasis (9%). Endoscopically, an obstructive endoluminal mass was found in all cases, and the foreign body was detected immediately in only 45% of cases. This type of presentation, suggestive of lung cancer, is frequently reported in endobronchial actinomycosis not associated with foreign body.11 It therefore seems important to perform follow-up bronchoscopy after antibiotic therapy to exclude the presence of a foreign body. Actinomycosis infection was diagnosed on bronchial biopsies in all cases. The presence of sulfur granules in biopsy samples is highly suggestive of actinomycosis.12 In CHEST / 121 / 6 / JUNE, 2002
2071
contrast, culture findings are usually negative, as A israelii is a strict anaerobe and is frequently associated with nonanaerobic contaminants. Bronchial aspiration samples may also be contaminated by oral flora. A israelii is usually sensitive to penicillins. Other effective antibiotic families are tetracyclines, lincosamides, and trimethoprim-sulfamethoxazole.12 The optimal duration of treatment has not been clearly established, but treatment for at least 45 days seems to be necessary in the case of thoracic actinomycosis.1 Our patients were treated with penicillin G or ampicillin, but one patient allergic to -lactam antibiotics was treated with a macrolide. Clindamycin can be an alternative therapy in these situations. Bronchoscopic removal of the foreign body was effective in all cases. However, in 45% of cases, the foreign body was only detected some time after starting antibiotics. Extraction procedures required bronchial aspiration (3 of 11 cases), biopsy forceps (6 of 11 cases), YAG laser (1 of 11 cases) or cryotherapy (1 of 11 cases). This review of the literature concerning endobronchial actinomycosis associated with foreign body aspiration confirms the development of this infection in debilitated patients with poor oral hygiene, frequently mimicking lung cancer. Endobronchial actinomycosis is usually diagnosed on histologic examination of bronchial biopsies and requires investigation of an associated bronchial foreign body, which may be missed on the initial assessment.
References 1 Hsieh MJ, Liu HP, Chang JP, et al. Thoracic actinomycosis. Chest 1993; 104:366 –370 2 Dalhoff K, Wallner S, Finck C, et al. Endobronchial actinomycosis. Eur Respir J 1994; 7:1189 –1191 3 McHardy G, Browne DC. Primary bronchial actinomycosis. South Med J 1943; 36:674 – 676 4 Umeki S, Nakajima M, Tsukiyama K, et al. Foreign bodyinduced bronchial actinomycosis with severe stenosis that must be distinguished from lung cancer. Nihon Kyobu Shikkan Gakkai Zasshi 1990; 28:481– 486 5 Mingrone H, Perrone R, La Rosa S, et al. Primary bronchial actinomycosis and foreign body. Medicina (B Aires) 1995; 55:337–340 6 Fernandez Jorge MA, Castro Villamor MA, Bartolome de Castro EM, et al. Thoracic actinomycosis and intrabronchial foreign body: presentation of two cases. An Med Interna 1995; 12:79 – 81 7 Julia G, Rodriguez de Castro F, Caminero J, et al. Endobronchial actinomycosis associated with a foreign body. Respiration 1991; 58:229 –230 8 Dicpinigaitis PV, Bleiweiss IJ, Krellenstein DJ, et al. Primary endobronchial actinomycosis in association with foreign body aspiration. Chest 1992; 101:283–285 9 Mignon F, Mesurolle B, Chambellan A, et al. Granulomatous reaction to a foreign body mimicking bronchogenic tumor: CT findings with virtual endoscopy. J Radiol 1997; 78:1181– 1184 10 Lan RS. Non-asphyxiating tracheobronchial foreign bodies in adults. Eur Respir J 1994; 7:510 –514 11 Ariel I, Breuer R, Kamal NS, et al. Endobronchial actinomycosis simulating bronchogenic carcinoma. Chest 1991; 99: 493– 495 12 Brown JR. Human actinomycosis: a study of 181 subjects. Hum Pathol 1973; 4:319 –330 2072
Progression of Idiopathic Pulmonary Fibrosis in Native Lungs After Single Lung Transplantation* Momen M. Wahidi, MD; James Ravenel, MD†; Scott M. Palmer, MD, MHS, FCCP; and H. Page McAdams, MD
This retrospective, single-center study was conducted to assess the response of native idiopathic pulmonary fibrosis (IPF) lungs to a potent cyclosporine-based immunosuppressive regimen in single-lung transplantation recipients. The study included IPF patients who had undergone single-lung transplantation and had chest CT scans before and after transplantation. Five patients underwent single-lung transplantation for IPF between April 1992 and January 2001, and met entry criteria. All patients were placed on an immunosuppressive regimen consisting of prednisone, azathioprine, and cyclosporine. In two of the five patients, ground glass attenuation in the native IPF lung improved post-transplantation. However, fibrotic changes progressed in all five patients. In patients with advanced IPF, a potent cyclosporine-based immunosuppressive regimen is not likely to have an effect on the progression of the disease. (CHEST 2002; 121:2072–2076) Key words: cyclosporine; idiopathic pulmonary fibrosis; lung transplantation Abbreviations: HRCT ⫽ high-resolution CT; IPF ⫽ idiopathic pulmonary fibrosis
pulmonary fibrosis (IPF) is an incurable I diopathic disease with a 5-year survival rate of 30 to 50% from the
time of diagnosis.1 To date, there are no sufficient data to support a favorable effect of any treatment on the progressive course of IPF. Corticosteroids have become the conventional treatment despite their limited efficacy.2 It is estimated that 15 to 30% of patients with IPF improve while receiving corticosteroids.3–5 Azathioprine has been used as a second-line therapeutic agent in IPF patients with poor responses or intolerance to corticosteroids. In one study, Raghu et al6 randomized patients to prednisone and azathioprine or to prednisone and placebo, and found a trend toward an improved survival rate in the combinedtreatment group. Several case reports7–10 have documented an encouraging response to cyclosporine in IPF patients. *From the Department of Internal Medicine (Drs. Wahidi and Palmer), Division of Pulmonary and Critical Care, and the Department of Radiology (Drs. Ravenel and McAdams), Duke University Medical Center, Durham, NC. †Now at the Medical University of South Carolina, Columbia, SC. Manuscript received September 11, 2001; revision accepted January 15, 2002. Correspondence to: Momen M. Wahidi, MD, Division of Pulmonary and Critical Care, Duke University Medical Center, Box 3221, Durham, NC 27710; e-mail: [email protected] Selected Reports