==== Front Int J Surg Case RepInt J Surg Case RepInternational Journal of Surgery Case Reports2210-2612Elsevier S2210-2612(18)30279-710.1016/j.ijscr.2018.07.029ArticleSurgical approach to a mycotic aneurysm of the pulmonary artery presenting with hemoptysis – A case report and a review of the literature Benhassen Leila Louise benhassen@live.dka⁎Højsgaard Anette aAllan Terp Kim ade Paoli Frank Vincenzo aba Department of Cardiothoracic and Vascular Surgery, Aarhus University Hospital, Palle-Juul Jensens Boulevard 99, DK-8200, Aarhus N, Denmarkb Department of Biomedicine, Aarhus University, Vennelyst Boulevard 4, DK-8000, Aarhus C, Denmark⁎ Corresponding author. benhassen@live.dk31 7 2018 2018 31 7 2018 50 92 96 25 5 2018 16 7 2018 24 7 2018 © 2018 The Author(s)2018This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).Highlights • Mycotic aneurysms of the pulmonary arteries are very rare with high mortality. • Risk groups are IV drug users and patients with congenital heart disorders. • Symptoms are often non-specific and may cause a diagnostic delay. • When hemoptysis is present, the suspicion of a pulmonary aneurysm should be raised. • For rapidly progressing proximal aneurysms, a midline approach is recommended. Introduction Mycotic aneurysms of the pulmonary arteries are very rare and have high mortality. Risk groups are intravenous drug users and patients with congenital heart disorders. The surgical approach varies due to a limited number of reported cases. Presentation of case We present a case of a mycotic aneurysm of the right pulmonary artery in a 56-year old man presenting with recurrent pneumonias, weight loss and hemoptysis. Discussion There is often a diagnostic delay because of non-specific symptoms mimicking more common disorders. Treatment strategies include conservative management, surgery and endovascular treatment. Conclusion This report demonstrates a rare case of aneurysm of the pulmonary artery presenting with hemoptysis. For rapidly progressing proximal aneurysms of the pulmonary arteries, the midline surgical approach is recommended. Keywords Case reportAneurysmPulmonary arteryHemoptysisMidline surgical approach ==== Body 1 Introduction Aneurysms are irreversible vascular dilatations involving all three layers of the vessel wall [1]. Infections causing aneurysms, mycotic aneurysms, are estimated to represent only 1–3% of all arterial aneurysms and are rarely located in pulmonary arteries [2]. Patients may present with dyspnea, chest pain, cough, pulmonary hypertension, right ventricular failure and hemoptysis. Hemoptysis is estimated to be present in less than 10% of cases, however, when present the suspicion of a pulmonary aneurysm should be raised [1,2]. The non-specific presentation often causes diagnostic delay resulting in critical progression of the aneurysm. We report here a case of successful surgical treatment for a mycotic aneurysm of the right pulmonary artery. The present case has been reported in line with the SCARE-criteria [3]. 2 Presentation of case A 56-year old man was admitted with recurrent pneumonias and symptoms of dyspnea and productive coughing for eight months, as well as hemoptysis and relapsing fever for the last three months. Moreover, he had a weight loss of 12 kilos, excessive tiredness and persistently elevated infection parameters. A PET/CT scan showed pneumonic infiltrates in the upper and lower lobe of the right lung and an enlarged mass in the right hilum, interpreted as lymph nodes. Bronchoscopy and bronchoalveolar lavage revealed no signs of malignancy. The patient was treated with antibiotics but showed no signs of recovery during the following three months. An additional CT scan with contrast was performed suggesting the enlarged mass in the right hilum to be an aneurysmatic expansion of the right pulmonary artery (55 mm) indicating a mycotic aneurysm-formation with peripheral pulmonary embolisms to all three lobes of the right lung (Fig. 1, Fig. 2). Following a multidisciplinary team meeting it was decided to manage the aneurysm conservatively with anticoagulants and antibiotics and repeat CT scan after 4 weeks. However, four days later an x-ray raised suspicion of progression of the aneurysm. An urgent CT scan confirmed progression of the aneurysm to 595 mm, as well as progression of pulmonary embolisms (Fig. 3). Transesophageal echocardiography (TEE) showed no signs of endocarditis or pulmonary hypertension. There was no confirmation of positive blood cultures. The treatment strategy was changed to a pneumonectomy of the right side.Fig. 1 1) Cross-sectional CT with contrast. Arrow pointing at the mycotic aneurysm with thrombus-formation located on the right pulmonary artery; 2) Longitudinal CT contrast. Arrow pointing at the mycotic aneurysm. Fig. 1Fig. 2 CT reconstruction from a posterior-lateral view. Arrow pointing at the aneurysm on the right pulmonary artery. Fig. 2Fig. 3 1) CT with contrast showing a mycotic aneurysm of the right pulmonary artery measuring 54.6 mm; 2) CT with contrast showing progression of the mycotic aneurysm measuring 59.5 mm. Fig. 3 A pneumonectomy of the right lung was performed through a median sternotomy. Intraoperatively, the tissue of the aneurysm was considered of good quality, which made it possible to resect the tissue in close proximity to the aneurysm. The operation was performed without complications and the patient made an uneventful recovery and was discharged 15 days after surgery. Biopsies from the aneurysm revealed no microbiological focus, despite this the patient received Carbapenem for two weeks and Moxifloxacin for another two weeks postoperatively. Four months after the surgery, the patient revealed no symptoms or signs of infection and has started working again. Control CT scan shows no abscesses or new aneurysm formation. 3 Discussion The case presented several diagnostic and management challenges. The enlarged mass in the right hilum found on X-ray was initially interpreted as enlarged lymph nodes. Due to malignancy-suspect symptoms such as weight loss, coughing and recurrent pneumonias, cancer was considered as a differential diagnosis, but was excluded on the subsequent PET/CT scan. Bronchoscopy and bronchoalveolar lavage revealed no signs of malignancy, and the patient was discharged with antibiotics. When the patient showed no signs of recovery after months, a CT scan was performed, and what was thought to be enlarged lymph nodes was now recognized as a pulmonary artery aneurysm with several septic thromboembolisms. Relative few cases have been reported of aneurysms of the pulmonary arteries. Known risk factors are IV drug use, congenital heart disease such as ventricular septal defect (VSD), generalized vasculitis, pulmonary hypertension, infection and immunosuppression [1]. Additional, according to Table 1 endocarditis seems to a major predisposing factor for development of mycotic aneurysms. Mycotic aneurysms of the pulmonary arteries constitute a minority of the aneurysms at this site, and as seen in Table 1 they are commonly caused by suppurativa bacterial infections such as staphylococci and streptococci or fungi including Candida and Aspergilli [4,5]. In the present case neither a bacteriological focus nor any of the risk factors mentioned above were present, however, since several septic thromboembolisms were located in the right lung, a mycotic aneurysm was a plausible diagnosis.Table 1 Clinical and laboratory features of reported patients with pulmonary mycotic artery aneurysm. Table 1Source, year Sex Age (y) Predisposing factor Systolic pulmonary arterial pressure (mmHg) Hemoptysis Infecting organisms Surgical treatment Survival Pirani, 1949 [7] M 10 Lymphoblastoma NS + Negative None - Lewisohn, 1957 [8] M 64 Pulmonary endocarditis NS - M. Tetragenus, S. Aureus, pneumococcus, proteus, Strep. Viridans None - Goh, 1974 [9] F 13 PDA, endocarditis 48 - S. Pyogenes Aneurysmectomy, closure of PDA + Goh, 1974 [9] M 7 PDA, endocaditis 58 - Negative None - Gorodezky, 1974 [10] F 18 PDA, endocarditis NS + Streptococcus None - Singer-Jordan, 1980 [11] M 23 Ventriculoatrial shunt NS + Propionibacterium acnes Pneumonectomy + Gelfand, 1981 [12] M 23 Drug abuse, ventriculoatrial shunt due to hydrocephalus NS + Propioni organisms, S. Epidermidis, Strep. Viridans Pneumonectomy + Choyke, 1982 [13] F 12 Corrected tetralogy of Fallot, tricuspid endocarditis NS - CNS, Aspergillus Tricuspid valve replacement - SanDretto [14] F 36 Drug abuse, endocarditis NS - CNS None + Navarro, 1984 [15] F 34 Drug abuse NS - NS None - Navarro, 1984 [15] M 33 Drug abuse, tricuspid endocarditis NS - S. Aureus, Streptcoccus, Kkebsiella, Diphtheroids None - Navarro, 1984 [15] F 27 Drug abuse NS - S. Aureus None - Caplin, 1985 [16] F 20 Closure of PDA NS - S. Aureus Closure of PDA, ligation of aneurysm + Morgan, 1986 [17] M 20 Drug abuse NS + S. Aureus None - Roush, 1988 [18] M 62 Tricuspid endocarditis NS - S. Aureus, C. Albicans Tricspid valve replacement - Vargas-Barron, 1992 [19] M 11 PDA, coarctation, vegetations on pulmonary artery NS - S. Aureus Correction of coarctation, closure of PDA, aneurysmectomy + Chung, 1995 [20] F 58 None NS - None None + Benveniste, 1998 [21] F 28 HIV, tricuspid and pulmonary endocarditis, drug abuse 60 + S. Aureus and Strep. Oralis Lobectomy - McLean, 1998 [6] M 28 Drug abuse NS - S. Aureus None + Lawrenson, 1999 [22] F 2 Tetralogy of Fallot, Blalock Taussig shunt, pulmonary endocarditis NS - Strep. Viridans Pulmonary valve replacement + Lertsapcharoen, 2002 [23] F 9 PDA, endocarditis NS + Streptococcus Closure of PDA + Dransfield, 2003 [24] F 49 None NS - Str. viridans None - Bozkurt, 2003 [25] F 6 VSD, tricuspid endocarditis NS - S. aureus Lobectomy, plication of aneurysm, closure of VSD, removal of vegetations of tricuspid valve + Kim, 2004 [5] M 71 None NS + Actinomyces Coil embolization - Greillier, 2005 [26] M 53 Acute myeloid leukemia NS - Aspergillus Fumigatus None - Saida, 2007 [27] F 49 Infection of pacemaker NS - S. Aureus None + Takao, 2007 [28] F 21 VSD, endocarditis NS - NS Coil embolization + Wilson, 2008 [29] F 22 Drug abuse, pulmonary endocarditis NS - S. Aureus None + Sever, 2010 [30] M 49 Hairy cell leukemia, dental abscess NS + Aspergillus Lobectomy - Al Banna, 2011 [4] M 48 Pericardial tamponade 27 - C. Albicans None + Groner, 2012 [31] M 4 DiGeorge syndrome with operated tetralogy of Fallot, xenograft endocarditis NS - C. Hominis Coil embolization - Papaioannou, 2014 [2] F 23 Drug abuse NS + S. Aureus Lobectomy + Toganel, 2014 [32] M 2 months Previous CVK, endocarditis NS + C. Lusitaniae Removal of thrombus in right ventricular outflow tract + Luo, 2015 [33] F 29 PDA, pulmonary endocarditis NS - NS Closure of PDA + Calais, 2017 [34] M 36 Endocarditis after Ross procedure NS - NS Pulmonary valve replacement + Knowles, 2017 [35] F 16 Operated Tetralogy of Fallot, endocarditis NS - S. Aureus Surgical revision of conduit + Piracha, 2018 [36] M 68 Chronic right upper lobe cavity NS + Negative Coil embolization - Srinivasa, 2018 [37] F 13 DiGeorge syndrome, repair of truncus arteriosus, pulmonary artery hypoplasia, endocarditis NS - Stapfylococcus Balloon occlusion - PDA: patent ductus arteriosus, VSD: ventricular septal defect, CVK: central venous catheter, NS: not speficied, CNS: coagulase-negative staphylococcus. A search of the PubMed database was undertaken on 05/07/18 with the search terms (“Aneurysm, Infected” [Mesh] AND “Pulmonary Artery”[Mesh]. 89 articles were returned. Articles in non-english, articles regarding pseudoaneurysms and non-relevant articles were excluded, which left 40 articles. Of these, 13 were unable to be accessed. The remaining 27 articles were included in our paper. Furthermore, 8 articles were found via references, which resulted in a total of 35 papers included in the review. The proposed pathological mechanisms are i) direct spreading to a pulmonary artery from an adjacent focus of pulmonary infection, ii) ischemic injury to the pulmonary arterial wall as a result of infection of the vasa vasorum or iii) direct extension into a vessel wall from an intraluminal septic thromboembolus or from hematogenous spreading [1,4]. According to Table 1, the latter is the most frequent route of transmission with endovascular seeding originating from endocarditis, IV drug use, skin abscesses or pneumonia [1]. Since TEE showed no signs of endocarditis and all blood cultures were negative, in our case direct spread from a pulmonary focus or endovascular seeding from the pulmonary embolisms are the most likely pathological mechanisms. Pulmonary angiography was previously gold standard, but recently MRI and CT with contrast have become the preferred diagnostic alternatives [4], which is confirmed in this case report. According to Bartter et al. the prognosis for mycotic aneurysms of the pulmonary arteries without interventional treatment is awful with mortality rates of 40–82% due to rupture [1]. Indeed, these mortality rates are confirmed in Table 1 showing a 63% (10 of 16 cases) mortality rate for patients not receiving surgical intervention, in contrast, patients receiving open surgical intervention with aneurysmectomy or removal of the affected part of the lung have a reduced mortality rate of 22% (2 of 9 cases). The management of these patients is difficult due to a lack of clear guidelines and sparse clinical experience. The proposed treatment strategies are diverse and include surgical interventions such as aneurysmectomy, lobectomy, pneumonectomy or embolotherapy [4,5]. Small mycotic aneurysms can also be treated conservatively, but regular CT scans are mandatory [6]. Less invasive approaches, e.g. lobectomy or segmental resection of lung parenchyma through a thoracotomy, are normally recommended, if feasible. However, for rapid progressing proximal aneurysms of the pulmonary artery, a midline approach through a sternotomy is to be recommended due to the possibility of central control in case of complications such as bleeding and the possibility to divide the structures more proximally in case of fragile tissue. Alternative nonsurgical interventional procedures, such as coil embolization or occlusion by detachable balloons might be used for peripheral pulmonary aneurysms, multiple bilateral pulmonary aneurysms or in patients who may not tolerate surgery [6]. In our case, the midline approach was chosen due to the central location and the rapid progression of the aneurysm, which indicated an unstable vessel wall with increased risk of hemorrhage. According to the literature the presence of hemoptysis appears to be a significant marker of instability of the lesion and a strong indicator of prompt intervention [1]. 4 Conclusion The diagnosis should be suspected when patients present with hemoptysis or signs of pulmonary hypertension, but also in the presence of more vague symptoms, especially in specific patient groups. The presence of hemoptysis and progression of the mycotic aneurysm should alert the clinician to consider a more aggressive treatment like aneurysmectomy or removal of the affected part of the lung. A midline approach is recommended in case of rapidly developing proximal aneurysms of the pulmonary artery to avoid potential complication of catastrophic bleeding and enable better surgical control. Conflict of interest No conflict of interest declared. Funding No funding was received for the study. Ethical approval Consent from the patient is sufficient and ethical approval is not needed in Denmark. Consent Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request. Author contribution All authors dr. Terp, dr. Højsgaard, dr. de Paoli and dr. Benhassen have taken part in conception of the study, drafting and revising the whole manuscript critically. All authors have given their final approval of the manuscript upon submission. Registration of research studies None. Guarantor Leila Louise Benhassen. ==== Refs References 1 Bartter T. Irwin R.S. Nash G. Aneurysms of the pulmonary arteries Chest 94 1988 1065 1075 3053058 2 Papaioannou V. Mikroulis D. Chrysafis I. Fotakis S. Pneumatikos I. Hemoptysis due to a mycotic pulmonary artery aneurism in an injecting drug user Thorac. Cardiovasc. Surg. 62 2014 453 455 23250844 3 Agha R.A. Fowler A.J. Saetta A. Barai I. Rajmohan S. Orgill D.P. for the SCARE Group The SCARE statement: consensus-based surgical case report guidelines Int. J. Surg. 34 2016 180 186 27613565 4 Al Banna R. Husain A. Suppurative pericarditis complicated by mycotic aneurysm of the pulmonary artery BMJ Case Rep. 2011 5 Kim H.S. Oh Y.W. Noh H.J. Lee K.Y. Kang E.Y. Lee S.Y. Mycotic pulmonary artery aneurysm as an unusual complication of thoracic actinomycosis Korean J. Radiol. 5 1 2004 68 71 15064561 6 McLean L. Sharma S. Maycher B. Mycotic pulmonary arterial aneurysms in an intravenous drug user Can. Respir. J. 5 4 1998 307 311 9753530 7 Pirani C.L. Ewart F.E. Jr Wilson A.L. Thromboendarteritis with multiple mycotic aneurysms of branches of the pulmonary artery Am. J. Dis. Child. 77 4 1949 460 473 18119705 8 Lewisohn M. Micrococcus tetragenus septicemia; report of a case including subacute bacterial endocarditis of the pulmonic valve and mycotic aneurysm, with autopsy findings and review of the literature AMA Arch. Intern. Med. 99 5 1957 824 832 13410188 9 Goh T.H. Mycotic aneurysm of the pulmonary artery. A report of 2 cases Br. Heart J. 36 4 1974 387 390 4407854 10 Gorodezky M. Maroto J.M. Mota J. Cardenas M. Contreras R. Mycotic aneurysms of the pulmonary artery Chest 66 2 1974 214 216 4408319 11 Singer-Jordan J.R. Mycotic aneurysm of the pulmonary artery complicating ventriculoatrial shunt AJR Am. J. Roentgenol. 135 2 1980 378 380 6773348 12 Gelfand E.T. Callaghan J.C. Mycotic pulmonary artery aneurysm: an unusual complication of ventriculo-atrial shunt Cardiovasc. Dis. 8 2 1981 271 275 15216218 13 Choyke P.L. Edmonds P.R. Markowitz R.I. Kleinman C.S. Laks H. Mycotic pulmonary artery aneurysm: complication of Aspergillus endocarditis AJR Am. J. Roentgenol. 138 6 1982 1172 1175 6979223 14 SanDretto M.A. Scanlon G.T. Multiple mycotic pulmonary artery aneurysms secondary to intravenous drug abuse AJR Am. J. Roentgenol. 142 1 1984 89 90 6606970 15 Navarro C. Dickinson P.C. Kondlapoodi P. Hagstrom J.W. Mycotic aneurysms of the pulmonary arteries in intravenous drug addicts. Report of three cases and review of the literature Am. J. Med. 76 6 1984 1124 1131 6547273 16 Caplin J.L. Dymond D.S. Barrett D.S. Rees R.S. Rees G.M. Spurrell R.A. Pulmonary mycotic aneurysms secondary to infective endocarditis in a patient with a persistent ductus arteriosus and partial anomalous pulmonary venous drainage Eur. Heart J. 6 11 1985 985 988 3841064 17 Morgan J.M. Morgan A.D. Addis B. Bradley G.W. Spiro S.G. Fatal haemorrhage from mycotic aneurysms of the pulmonary artery Thorax 41 1 1986 70 71 3754657 18 Roush K. Scala-Barnett D.M. Donabedian H. Freimer E.H. Rupture of a pulmonary artery mycotic aneurysm associated with candidal endocarditis Am. J. Med. 84 1 1988 142 144 3337118 19 Vargas-Barron J. Avila-Rosales L. Romero-Cardenas A. Rijlaarsdam M. Keirns C. Buendia A. Echocardiographic diagnosis of a mycotic aneurysm of the main pulmonary artery and patent ductus arteriosus Am. Heart J. 123 6 1992 1707 1709 1595557 20 Chung C.W. Doherty J.U. Kotler R. Finkelstein A. Dresdale A. Pulmonary artery aneurysm presenting as a lung mass Chest 108 4 1995 1164 1166 7555134 21 Benveniste O. Bruneel F. Bédos J.P. Wolff M. Lesèche G. Leport C. Ruptured mycotic pulmonary artery aneurysm: an unusual complication of right-sided endocarditis Scand. J. Infect. Dis. 30 6 1998 626 629 10225401 22 Lawrenson J. Stirling J. Hewitson J. Images in cardiology: mycotic aneurysm of the left pulmonary artery in a child with tetralogy of Fallot and Streptococcus viridans infective endocarditis Heart 82 1 1999 88 10377315 23 Lertsapcharoen P. Chottivittayatarakorn P. Benjacholamas V. Mycotic aneurysms of the pulmonary arteries Heart 88 5 2002 524 12381650 24 Dransfield M.T. Johnson J.E. A mycotic pulmonary artery aneurysm presenting as an endobronchial mass Chest 124 4 2003 1610 1612 14555599 25 Bozkurt A.K. Oztunç F. Akman C. Kurugoglu S. Eroğlu A.G. Multiple pulmonary artery aneurysms due to infective endocarditis Ann. Thorac. Surg. 75 2 2003 593 596 12607688 26 Greillier L. Barlesi F. Fraticelli A. Gimenez C. Chetaille B. Gaubert J.Y. Fatal pulmonary haemorrhage from a mycotic pulmonary artery aneurysm Int. J. Tuberc. Lung Dis. 9 6 2005 702 703 15971404 27 Saida T. Ninomiya H. Yamasaki M. Yamauchi T. Mycotic pulmonary artery aneurysm as a late complication of infective endocarditis after pacemaker implantation Radiat. Med. 25 5 2007 240 242 17581714 28 Takao H. Makita K. Doi I. Watanabe T. Mycotic pulmonary artery aneurysm diagnosed 18 years after bacterial endocarditis Clin. Imaging 31 2 2007 131 133 17320781 29 Wilson T.N. Tew K. Taranath A. Multiple mycotic aneurysms of the pulmonary arteries resolving with conservative management: multislice CT examination findings J. Thorac. Imaging 23 3 2008 197 201 18728549 30 Sever M. Verstovsek S. Erasmus J. Jr. Mattiuzzi G.N. Mycotic pulmonary artery aneurysm due to Aspergillus infection in a patient with leukemia: case report and review of the literature Leuk. Res. 34 5 2010 e133 e136 20045559 31 Groner A. Kowalsky S. Arnon R. Tosi M.F. Endocarditis due to cardiobacterium hominis in a 4-year-old boy, complicated by right lower lobe pulmonary artery mycotic aneurysm J. Pediatric Infect. Dis. Soc. 2 3 2013 278 280 26619483 32 Toganel R. Benedek T. Benedek I. Giant mycotic pulmonary artery aneurysms in a newborn Eur. Heart J. Cardiovasc. Imaging 15 8 2014 885 24574453 33 Luo T. Jiang L. Peripheral mycotic pulmonary artery aneurysms J. Card. Surg. 30 2 2015 170 172 25545130 34 Calais J. Pasi N. Nguyen V. Hyafil F. Mycotic aneurysm in a pulmonary artery detected with 18F-fluorodeoxyglucose positron emission tomography/computed tomography imaging Eur. Heart J. 38 46 2017 3474 28057774 35 Knowles K.A. Alvin M. Dunn E. Mycotic pulmonary artery aneurysm secondary to septic emboli BMJ Case Rep. 2017 December 2017 pii: bcr-2017-223439 36 Piracha S. Mahmood A. Qayyum N. Ganaie M.B. Massive haemoptysis secondary to mycotic pulmonary artery aneurysm in subacute invasive aspergillosis BMJ Case Rep. 2018 April 2018 pii: bcr-2017-223088 37 Srinivasa R.N. Srinivasa R.N. Zhao S. Gemmete J.J. Chick J.F.B. Balloon occlusion technique with ethylene vinyl alcohol for the treatment of a pediatric pulmonary artery mycotic aneurysm Radiol. Case Rep. 13 1 2017 121 124 29487647