Pulmonary actinomycosis: a case undergoing resection through video-assisted thoracic surgery (VATS)

Pulmonary actinomycosis: a case undergoing resection through video-assisted thoracic surgery (VATS) 1 first-author 2 3 corresp ‡Corresponding Author ‡Corresponding Author †E-mail:luh572001@yahoo.com.tw †E-mail:luh572001@yahoo.com.tw luh572001@yahoo.com.tw 4 4 Department of Medicine, Chia-Yi Christian Hospital, 600 Taichung, Taiwan, China Department of Medicine, Chia-Yi Christian Hospital, 600 Taichung, Taiwan, China Department of Pathology, Chung-Shan Medical University and Hospital, 402 Taichung, Taiwan, China Department of Pathology, Chung-Shan Medical University and Hospital, 402 Taichung, Taiwan, China Department of Surgery, Chung-Shan Medical University and Hospital, 402 Taichung, Taiwan, China Department of Surgery, Chung-Shan Medical University and Hospital, 402 Taichung, Taiwan, China Department of Medicine, Chung-Shan Medical University and Hospital, 402 Taichung, Taiwan, China Department of Medicine, Chung-Shan Medical University and Hospital, 402 Taichung, Taiwan, China Actinomycosis is an uncommon disease, which is usually manifested as cervicofacial infection and related to poor oral hygiene or compromised immune function. Pulmonary actinomycosis is rare, but its diagnosis is changing due to its variable presentation and the similarity in appearance to other intrapulmonary diseases. Here we report an 80-year-old man with a solitary pulmonary nodule over the left upper lobe. Pulmonary neoplasm was highly suspected in this patient and thus resection of the mass was undertaken through video-assisted thoracic surgery (VATS). Histopathological examination demonstrated this patient had an Actinomyeces infection. While the application of VATS in patients with pulmonary actinomycosis has rarely been reported in literature, we conclude that VATS is valuable for the diagnosis and treatment of patients with undetermined pulmonary nodule(s). Pulmonary actinomycosis Video-assisted thoracic surgery (VATS) Resection INTRODUCTION 1 Actinomyces spp., first described in the 19th century, exist in the soil and oral cavity, and the species name was derived from its ray-like arrangement with sulfur granules (Rippon, 1988 1988 ). They can infect human beings as well as other animals (Bennhoff, 1984 1984 ; Reichel and Wragg, 2007 2007 ). Actinomycosis is uncommon, and usually occurs in patients with compromised immune function or poor oral hygiene (Kinnear and MacFarlane, 1990 1990 ). It usually results in cervicofacial infection, and pulmonary infection is relatively rare (Schaal and Lee, 1992 1992 ). In this study, a patient with pulmonary actinomycosis mimicking lung tumor who underwent a pulmonary resection through video-assisted thoracic surgery (VATS) was reported. CASE REPORT 1 An 80-year-old man was admitted with the complaints of intermittent cough with blood-tinged sputum for 3 months. His cough usually aggravated at night, and no prominent chest pain, anorexia or body weight loss was noted. The medicine he took from other clinic did not markedly improve the symptoms. He smoked cigarettes 1 pack a day for over 50 years and quitted 3 years ago. He had a gingival inflammation several months ago and underwent local treatment irregularly. A physical examination showed that this patient was moderately developed and nourished and had clear consciousness. Respiratory examinations found no cervical lymphadenopathy, musculoskeletal disorder or other abnormalities. His blood pressure was 150/80 mmHg. Chest X-ray revealed a solitary nodular shadow over the left upper lung field (Fig. 1 1 ). The computed tomogram (CT) showed a speculated mass, 2 cm in diameter, over the left B1-2 segment of the left lung, which was highly suspected as a malignancy (Fig. 2 2 ). Therefore he was referred to the thoracic surgical ward for a further examination and treatment. In our ward, the positron emitted tomogram (PET) revealed a hypermetabolic lesion over the left upper lobe of the lung of the patient, which favors a malignancy. However, cytology of sputum and transbronchial brushing were negative for malignant cells, along with the normal bronchoscope examination and negative for bacterial, tuberculous or fungal infection from the repeated sputum cultural results. A CT guided biopsy was recommended but the patient refused to undergo this procedure. The patient underwent a surgical resection through the VATS. The B1 segmentectomy was performed smoothly through a 4 cm utility incision and a 1 cm port for scope. The gross picture of the mass is yellowish, elastic on palpation, and without prominent pleural retraction (Fig. 3 3 ). The frozen section did not show malignant cells and the subsequent pathological examination demonstrated the aggregates of filamentous Gram-positive microorganism in the characteristic of “sulfur granules”, indicating actinomycosis (Fig. 4 4 ). This patient was discharged uneventfully and underwent penicillin p.o. treatment at our clinic for 2 months. He is alive and well now and has had a regular follow-up for 6 months. DISCUSSION 1 Actinomyes spp., belonging to a family of Gram-positive and anaerobic bacteria and existing on the soil, are normally found in human organs such as the oropharynx, gastrointestinal tract and female genitalia (Smego and Foglia, 1998 1998 ). A . israelli is the most commonly subtype that causes human diseases (Weese and Smith, 1975 1975 ). Actinomycosis has been reported around the world for more than one hundred years, but the incidence of all forms of actinomycosis has remarkably declined in recent years, especially in the developed country (Russo, 1995 1995 ). Pulmonary involvement is rare, accounting for approximately 15% of all patients with actinomycosis (Mabeza and MacFarlane, 2003 2003 ). The pathogenesis of actinomycosis is still unclear. Most cases with actinomycosis are associated with a variety of other microorganisms (Holm, 1950 1950 ). Clinical infection is usually related to the disruption of the mucosal barrier that allows the organism to invade, such as dental sepsis, appendicitis, diverticulitis, surgery, or intrauterine devices (Miller and Haddad, 1998 1998 ; Lippes, 1999 1999 ). Pulmonary actinomycosis might be related to poor oral hygiene or aspiration of gastrointestinal fluid, and as well the immuno-compromised status (Heffner, 1988 1988 ; Klapholz et al., 1989 1989 ). Radiographic pictures of pulmonary actinomycosis varied and could mimic a wide spectrum of benign and malignant diseases (Conant and Wechsler, 1992 1992 ). The chest X-rays range from small nodular lesion to cavitating lesions with pleural, chest wall, transdiaphragmatic, or spine involvement (Flynn and Felson, 1970 1970 ). Actinomycosis has the characteristic to penetrate the tissue plane, resulting in fistula or abscess formation. The CT findings of patients with pulmonary actinomycosis include patchy, nodular, or cavitating pulmonary lesions, as well as pleural or mediastinal lesions (Allen et al., 1987 1987 ). The value of PET in the diagnosis of pulmonary actinomycosis is not clear (Hoekstra et al., 1999 1999 ). Up to 25% of cases with thoracic actinomycosis were initially misdiagnosed as malignancy (Mabeza and MacFarlane, 2003 2003 ; Moore and Scannell, 1968 1968 ). The diagnosis of thoracic actinomycosis remains a clinical challenge, not only because it is uncommon but also because the culture of this bacterium from the sputum or bronchoalveolar secretions is technically difficult (Wong et al., 2004 2004 ), and as well sometimes represents merely colonization (not pathological microorganism) (Ariel et al., 1991 1991 ). A reliable diagnosis of this pathogen still requires histological or microbiological examinations. Biopsy through fiberoptic bronchoscopy can be used in patients with endobronchial actinomycosis (Ariel et al., 1991 1991 ), and the ultrasound or CT guided biopsy used for the diagnosis of patients with peripheral lung lesions (Das, 1994 1994 ). Since the clinical and gross pictures of pulmonary actinomycosis intra-operatively obtained are indiscernible from that of carcinoma, a minimally invasive approach, such as VATS, as well as a limited pulmonary resection and a frozen section to decide the subsequent procedures, is recommended (Moore and Scannell, 1968 1968 ; Luh and Liu, 2006 2006 ). The application of VATS in the diagnosis and treatment of pulmonary actinomycosis, has only been reported twice in literature (Kobashi et al., 2004 2004 ; Kakuda et al., 1997 1997 ). Our case shows that VATS is more reliable than the CT or sono-guided biopsy for the diagnosis of pulmonary actinomycosis, and less invasive than an open thoracotomy for the treatment of patients with undetermined pulmonary nodule(s). Pathological features of pulmonary actinomycosis include a granulomatoid lesion with or without giant or epitheloid cells (Brown, 1973 1973 ). The appearance of radiating arranged sulfur granules is the pathological hallmark of this disease. However, these granules could be appeared in other diseases such as nocardiosis or chromomycosis as well (Brown, 1973 1973 ). The choice of antimicrobial agents against actinomycosis is penicillin, and the duration of a therapy depends on the severity of the disease and the efficacy of the treatment, usually lasting at least one to two months (Slade et al., 1973 1973 ). A recent study containing eight patients who underwent a 4-week course of imipenem-cilastain therapy reported a satisfactory result (Yew et al., 1999 1999 ). We conclude that the application of VATS is more reliable in the diagnosis, and less invasive in the treatment of patients with pulmonary actinomycosis manifested as undetermined pulmonary nodule(s). References HAIII Allen , JC Scatarige , MH Kim . Actinomycosis: CT findings in six patients . AJR , 1987 . 149 ( 6 ): 1255 - 1258 . 3500618 . Actinomycosis: CT findings in six patients I Ariel , R Breuer , NS Kamal , I Ben-Dov , P Mogle , E Rosenmann . Endobronchial actinomycosis simulating bronchogenic carcinoma: diagnosis by biopsy . Chest , 1991 . 99 ( 2 ): 493 - 495 . 1989815 . Endobronchial actinomycosis simulating bronchogenic carcinoma: diagnosis by biopsy DF Bennhoff . Actinomycosis: diagnostic and therapeutic considerations and review of 32 cases . Laryngoscope , 1984 . 94 ( 9 ): 1198 - 1217 . 6381942 DOI: 10.1288/00005537-198409000-00013 http://doi.org/10.1288/00005537-198409000-00013 . Actinomycosis: diagnostic and therapeutic considerations and review of 32 cases JR Brown . Human actinomycosis. A study of 181 subjects . Human Pathol , 1973 . 4 ( 3 ): 319 - 330 . . Human actinomycosis. A study of 181 subjects EF Conant , RJ Wechsler . Actinomycosis and nocardiosis of the lung . J Thorac Imaging , 1992 . 7 ( 4 ): 75 - 84 . 1404547 DOI: 10.1097/00005382-199209000-00010 http://doi.org/10.1097/00005382-199209000-00010 . Actinomycosis and nocardiosis of the lung DK Das . Actinomycosis in fine needle aspiration cytology . Cytopathology , 1994 . 5 ( 4 ): 243 - 250 . 7948761 DOI: 10.1111/j.1365-2303.1994.tb00426.x http://doi.org/10.1111/j.1365-2303.1994.tb00426.x . Actinomycosis in fine needle aspiration cytology MW Flynn , B Felson . The roentgen manifestations of thoracic actinomycosis . AJR , 1970 . 110 ( 4 ): 707 - 716 . . The roentgen manifestations of thoracic actinomycosis JE Heffner . Pleuropulmonary manifestations of actinomycosis and nocardiosis . Semin Respir Infect , 1988 . 3 ( 4 ): 352 - 361 . 3062727 . Pleuropulmonary manifestations of actinomycosis and nocardiosis CJ Hoekstra , OS Hoekstra , JP Teengs , PE Postmus , EF Smit . Thoracic actinomycosis imaging with fluorine-18 fluorodeoxyglucose positron emission tomography . Clin Nucl Med , 1999 . 24 ( 7 ): 529 - 530 . 10402013 DOI: 10.1097/00003072-199907000-00016 http://doi.org/10.1097/00003072-199907000-00016 . Thoracic actinomycosis imaging with fluorine-18 fluorodeoxyglucose positron emission tomography P Holm . Studies on the aetiology of human actinomycosis. I. The “other” microbes of actinomycosis and their importance . Acta Pathol Microbiol Scand , 1950 . 27 ( 3 ): 736 . Studies on the aetiology of human actinomycosis. I. The “other” microbes of actinomycosis and their importance J Kakuda , B Omari , R Renslo , R Scott , F Baumgartner . CT guided needle localization for video-thoracoscopic resection of pulmonary nodules . Eur J Med Res , 1997 . 2 ( 8 ): 340 - 342 . 9262486 . CT guided needle localization for video-thoracoscopic resection of pulmonary nodules WJ Kinnear , JT MacFarlane . A survey of thoracic actinomycosis . Respir Med , 1990 . 84 ( 1 ): 57 - 59 . 2371423 . A survey of thoracic actinomycosis A Klapholz , W Talavera , E Rorat , E Salsitz , C Widrow . Pulmonary actinomycosis and human immunodeficiency virus infection . Mt Sinai J Med , 1989 . 56 ( 4 ): 300 - 303 . 2797022 . Pulmonary actinomycosis and human immunodeficiency virus infection Y Kobashi , K Yoshida , N Miyashita , Y Niki , T Matsushima . Thoracic actinomycosis with mainly pleural involvement . J Infect Chemother , 2004 . 10 ( 3 ): 172 - 177 . 15290457 DOI: 10.1007/s10156-004-0310-x http://doi.org/10.1007/s10156-004-0310-x . Thoracic actinomycosis with mainly pleural involvement J Lippes . Pelvic actinomycosis: a review and preliminary look at prevalence . Am J Obstet Gynaecol , 1999 . 180 ( 2 ): 265 - 269 . DOI: 10.1016/S0002-9378(99)70198-5 http://doi.org/10.1016/S0002-9378(99)70198-5 . Pelvic actinomycosis: a review and preliminary look at prevalence SP Luh , HP Liu . Video-assisted thoracic surgery. The past, present status, and the future . J Zhejiang Univ Sci B , 2006 . 7 ( 2 ): 118 - 128 . 16421967 DOI: 10.1631/jzus.2006.B0118 http://doi.org/10.1631/jzus.2006.B0118 . Video-assisted thoracic surgery. The past, present status, and the future GF Mabeza , J MacFarlane . Pulmonary actinomycosis . Eur Respir J , 2003 . 21 ( 3 ): 545 - 551 . 12662015 . Pulmonary actinomycosis M Miller , AJ Haddad . Cervicofacial actinomycosis . Oral Surg Oral Med Oral Path Oral Radiol Endod , 1998 . 85 ( 5 ): 496 - 508 . DOI: 10.1016/S1079-2104(98)90280-3 http://doi.org/10.1016/S1079-2104(98)90280-3 . Cervicofacial actinomycosis WR Moore , JG Scannell . Pulmonary actinomycosis simulating cancer of the lung . J Thorac Cardiovasc Surg , 1968 . 55 ( 2 ): 193 - 195 . 5635978 . Pulmonary actinomycosis simulating cancer of the lung R Reichel , P Wragg . Isolation of actinomyces hyovaginalis from a lung lesion in a pig . Vet Rec , 2007 . 10 ( 6 ): 203 . Isolation of actinomyces hyovaginalis from a lung lesion in a pig JW Rippon . MJ Wonsiewicz . Medical Mycology . The Pathogenic Fungi and the Pathogenic Actinomycetes , 1988 . 3rd Ed : Philadelphia W.B. Saunders Co. , 30 - 52 . . Medical Mycology TA Russo . GL Mandell . Agents of Actinomycosis . Principles and Practice of Infectious Disease , 1995 . 5th Ed : New York Churchill Livingstone , 2645 - 2654 . . Agents of Actinomycosis KP Schaal , H Lee . Actinomycete infections in human—a review . Gene , 1992 . 115 ( 1-2 ): 201 - 211 . 1612438 DOI: 10.1016/0378-1119(92)90560-C http://doi.org/10.1016/0378-1119(92)90560-C . Actinomycete infections in human—a review PR Slade , BV Slesser , J Southgate . Thoracic actinomycosis . Thorax , 1973 . 28 ( 1 ): 73 - 85 . 4568119 . Thoracic actinomycosis RA Smego , G Foglia . Actinomycosis . Clin Infect Dis , 1998 . 26 ( 6 ): 1255 - 1261 . (quiz in p.1262-1263) 9636842 . Actinomycosis WC Weese , IM Smith . A study of 57 cases of actinomycosis over a 36-year period . Arch Intern Med , 1975 . 135 ( 12 ): 1562 - 1568 . 1200725 DOI: 10.1001/archinte.135.12.1562 http://doi.org/10.1001/archinte.135.12.1562 . A study of 57 cases of actinomycosis over a 36-year period RH Wong , AD Sihoe , KH Thung , IY Wan , MB Ip , AP Yim . Actinomycosis: an often forgotten diagnosis . Asian Cardiovasc Thorac Ann , 2004 . 12 ( 2 ): 165 - 167 . 15213087 . Actinomycosis: an often forgotten diagnosis WW Yew , PC Wong , J Lee , SL Fung , CF Wong , CY Chan . Report of eight cases of pulmonary actinomycosis and their treatment with imipenem-cilastatin . Monaldi Arch Chest Dis , 1999 . 54 ( 2 ): 126 - 129 . 10394825 . Report of eight cases of pulmonary actinomycosis and their treatment with imipenem-cilastatin 10.1631/jzus.2007.B0721 2007-07-16 2007-08-06 2022-01-05 Copyright © 2007, Journal of Zhejiang University Science 2007 Journal of Zhejiang University-SCIENCE B (Biomedicine and Biotechnology) 10 08