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 Table of Contents  
INTERESTING CASE REPORT
Year : 2021  |  Volume : 5  |  Issue : 1  |  Page : 47-49

Fatal Mycotic Pulmonary Artery Aneurysms in Patients with Congenital Heart Disease Complicated by Right-sided Infective Endocarditis: A Report of Two Cases


Department of Pediatric Cardiology, Madras Medical Mission, Chennai, Tamil Nadu, India

Date of Submission02-Apr-2020
Date of Decision04-May-2020
Date of Acceptance21-Jun-2020
Date of Web Publication05-Apr-2021

Correspondence Address:
Dr. Kothandam Sivakumar
Department of Pediatric Cardiology, Institute of Cardio Vascular Diseases, Madras Medical Mission, 4a, Dr. J J Nagar, Mogappair, Chennai - 600 037, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiae.jiae_15_20

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  Abstract 

Mycotic aneurysms of pulmonary arteries are potentially fatal conditions occurring in patients ith congenital heart disease complicated by infective endocarditis folloing surgery or interventions. Presentation is indolent and symptoms may be nonspecific. Unless suspected and investigated ith computed tomography, they are missed until their advanced stage. Treatment options are limited as shon in to patients presented in this report.

Keywords: Computed tomography, congenital heart disease, echocardiography, infective endocarditis, mycotic aneurysm, pulmonary hemorrhage


How to cite this article:
Palaparti R, Raj G R, Sivakumar K. Fatal Mycotic Pulmonary Artery Aneurysms in Patients with Congenital Heart Disease Complicated by Right-sided Infective Endocarditis: A Report of Two Cases. J Indian Acad Echocardiogr Cardiovasc Imaging 2021;5:47-9

How to cite this URL:
Palaparti R, Raj G R, Sivakumar K. Fatal Mycotic Pulmonary Artery Aneurysms in Patients with Congenital Heart Disease Complicated by Right-sided Infective Endocarditis: A Report of Two Cases. J Indian Acad Echocardiogr Cardiovasc Imaging [serial online] 2021 [cited 2021 Jul 23];5:47-9. Available from: https://www.jiaecho.org/text.asp?2021/5/1/47/313087


  Introduction Top


Mycotic aneurysms of pulmonary arteries caused by infections are rare, but carry high mortality due to nonspecific symptoms, delays in diagnosis, and difficulties in management.[1] They are life-threatening as they may lead to aspiration and asphyxia after intrapulmonary hemorrhage, dissection of pulmonary artery, and sudden death. Multimodality imaging remains the key to diagnose this rare condition, assess its progress, and plan the treatment.[2] To cases of fatal mycotic pulmonary artery aneurysms ith nonspecific presentations are reported to highlight the difficulties in diagnosis and management.


  Case Reports Top


Case 1

A 24-day-old male neonate eighing 3.3 kg ith no syndromic phenotype and no clinical clues toard immunocompromise as diagnosed to have supracardiac total anomalous pulmonary venous drainage through a right vertical vein to the junction of the right superior caval vein to the right atrium along ith hyperkinetic pulmonary hypertension. Surgical repair ith anastomosis of the posterior common venous chamber to the anterior left atrium as uneventful ith no postoperative residual narroing of the anastomosis on echocardiography and discharge from hospital on ninth postoperative day. His echocardiogram at 1-month follo-up did not reveal any abnormality. After an asymptomatic 2 months, recurrence of breathlessness and poor feeding led to a readmission, hen he eighed 3.9 kg. A repeat echocardiography shoed features of severe pulmonary arterial hypertension and depressed right ventricular contractility. There as a mild turbulence of flo in the pulmonary venous chamber anastomosis ith a mean gradient of 9 mmHg. A purulent discharge from subxiphoid area related to the exit of the pacing ire and blood cultures gre methicillin-sensitive Staphylococcus aureus. After initiating appropriate antibiotics, a detailed echocardiography revealed a retrosternal abscess cavity separating the sternum and the heart and vegetations on tricuspid valve, confirming a idespread infection [Figure 1] and [Video 1]. A large heterogeneous opacity in the right loer zone on chest X-ray and retrosternal abscess on echocardiography prompted a contrast-enhanced computed tomography. It shoed a retrosternal organized abscess and a large right pulmonary artery aneurysm ith surrounding consolidation of the right loer lobe [Figure 2]. A chest ound re-exploration and drainage of the retrosternal abscess as done through a limited sternotomy. Compromised general condition and unstable ventilatory parameters did not permit right pneumonectomy and forced a conservative strategy. A massive bleeding due to rupture of the aneurysm despite bronchial isolation ith a Fogarty balloon catheter through the endotracheal tube led to death due to hemorrhagic shock.
Figure 1: Modified echocardiographic four-chamber view. (a) Vegetation in tricuspid valve (single arrow), dilated right atrium and right ventricle, and retrosternal abscess (double arrow) in front of the right ventricle. (b) A low parasternal short axis view also demonstrates the retrosternal abscess. LA: Left atrium, LV: Left ventricle, RA: Right atrium, RV: Right ventricle

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Figure 2: Right lower pulmonary artery branch mycotic aneurysm (double arrow) is shown in posteroanterior view of chest X-ray (a), and axial (b) and coronal (c) computed tomographic slices. The retrosternal abscess from the epicardial pacing wire is shown by single arrow

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Video 1: Case 1 shoing the post-operative chest X-ray, echocardiography during the first follo-up, and during index presentation. Computed tomographic images of mycotic pulmonary artery aneurysm are also shon.

[Additional file 1]

Case 2

A 7-year-old boy ith pan-hypopituitarism on groth hormone, thyroid, and steroid supplements as diagnosed to have tetralogy of Fallot ith pulmonary atresia, underent right modified Blalock-Taussig shunt at 3-months and left modified Blalock-Taussig shunt after 2 years as the pulmonary arteries remained small. A catheter pulmonary angiogram at 7 years of age shoed hypoplastic bilateral pulmonary arteries, poor arborization to both upper lobes and stenosis of the right and left pulmonary arteries at shunt insertion sites. After a total surgical correction ith a hand-fashioned bovine pericardial conduit ith pericardial patch augmentation of both mediastinal pulmonary arteries, he as discharged ith adequate right ventricular function, moderate bilateral pulmonary artery gradients, and right ventricular systolic pressures of 60 mmHg. His re-evaluation 1 year after surgery shoed increasing right ventricular pressures to 95 mmHg and mild right ventricular dysfunction. After a three-dimensional reconstruction from a rotational angiogram, stent angioplasty of both pulmonary arteries reduced right ventricular pressures from 100 to 45 mmHg ithout any pulmonary artery gradients. His clinical revie and echocardiogram at 1 and 6 months after stenting did not reveal any abnormality. He presented ith cough and hemoptysis 10 months after the stent angioplasty. Echocardiogram shoed vegetations in pulmonary conduit causing a gradient of 100 mmHg. His chest X-ray shoed a ell-defined circular opacity arising from right pulmonary artery [Figure 3] and [Video 2]. Positron emission tomography shoed metabolically active bilobed aneurysm from the right loer lobe pulmonary artery branch [Figure 4]. Fluorodeoxyglucose uptake as noted in mediastinal, hilar, and interlobar lymph nodes also. Blood investigations shoed leukocytosis and elevated erythrocyte sedimentation rates, but cultures remained sterile. Treatment as initiated ith culture-negative endocarditis protocol, and empiric antituberculous drugs ere added due to chronic steroid use. His parents denied consent for a radical aneurysm surgery involving lobectomy through adhesions of the previous thoracotomy. A massive hemoptysis led to asphyxia, hemorrhagic shock, and demise.
Figure 3: Parasternal shor t axis echocardiogram. (a) Vegetation (single arrow) in the conduit; (b) a homogenous opacity (double arrows) in right lower zone of chest X-ray Indicates the mycotic aneurysm

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Figure 4: Axial slices of computed tomography at hilar (a) and lower levels (b) demonstrate bilateral pulmonary artery stents and large mycotic aneurysm in the right lower lobe pulmonary artery branch, which is highlighted well with surrounding consolidation (double arrow) on the coronal slice (c)

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Video 2: Case 2 shoing rotational angiogram of pulmonary arteries ith significant branch pulmonary artery stenosis, the final result after bilateral pulmonary artery stenting, Chest x-ray and echocardiography at 6 months of follo-up and during index presentation. Computed tomographic images of pulmonary artery aneurysm are also shon.

[Additional file 2]


  Discussion Top


Mycotic pulmonary artery aneurysm is a ell-defined entity and carries a mortality of 50%–100% despite aggressive management.[1] They have been diagnosed in various forms of congenital heart diseases folloing palliative or corrective surgeries.[3],[4],[5] And, intracardiac surgery is a common predisposing factor.[6] They result due to direct spread to a pulmonary artery from an adjacent focus of lung infection; ischemic injury to pulmonary artery all resulting from infection of vasa vasorum; or direct extension to vessel all from a septic thromboembolus.[7] While the former mechanism explains aneurysms complicating tuberculosis, aspergillosis, or actinomycosis, the last mechanism explains aneurysms folloing endocarditis as in our patient.[8] A strong association beteen mycotic aneurysms and pulmonary hypertension is also ell noted. It is thought that in a vessel all already under stress, septic emboli can lodge and cause aneurysm formation.[9]

In the first patient, the pathogenic organism that gre from the pacing ire site possibly led to indolent endocarditis and mycotic aneurysm that clinically remained silent for the to intervening months. As the clinical course remained uneventful, cultures ere not sent from the central line tips or pacing ires during the postoperative recovery. In the second patient, a long 2-year interval from surgery and 1-year interval folloing the stent angioplasty indicated that the infective inoculum did not enter the patient during these interventions. In addition to a high possibility of acquiring infections during surgery or catheter interventions, the chronic steroid-induced immunosuppression also played a role.

Mortality is loer in various noninfectious causes including vasculitis such as Behcet's syndrome and Hughes-Stovin syndrome, primary pulmonary hypertension, lung neoplasms, trauma, iatrogenic injury like chest tube placement and lung ablation, radiation therapy, and lung resection. Differentiation of mycotic aneurysms from congenital and noninfective causes is often obvious and changes management strategies. When cases present indolently, blood cultures, inflammatory markers, procalcitonin, and multimodality imaging guide in diagnosis. Hemoptysis is a sentinel and ominous sign. Computed tomography is the imaging modality of choice for the detection of pulmonary artery aneurysms ith high sensitivity and specificity.[2] Surgical treatment for mycotic aneurysms ith aneurysmorrhaphy or aneurysmectomy, lobectomy, and pneumonectomy carry high procedural risks, so are the various endovascular therapeutic modalities such as covered stent exclusion, balloon embolization, stent-assisted coil embolization, and glue embolization.[10] Unless suspected ith a lo threshold, the diagnosis is often delayed.


  Conclusion Top


Mycotic pulmonary artery aneurysm is a fatal sequelae of right-sided endocarditis, often complicates congenital heart diseases after surgery or interventions. Presentations may be indolent, nonspecific and hemoptysis is a serious arning sentinel sign. Diagnosis needs computed tomography often, but treatment options are limited by comorbidities of the patient.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials ill not be published and due efforts ill be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Park HS, Chamarthy MR, Lamus D, Saboo SS, Sutphin PD, Kalva SP. Pulmonary artery aneurysms: Diagnosis endovascular therapy. Cardiovasc Diagn Ther 2018;8:350-61.  Back to cited text no. 1
    
2.
Chen Y, Gilman MD, Humphrey KL, Salazar GM, Sharma A, Muniappan A, et al. Pulmonary Artery Pseudoaneurysms: Clinical Features and CT Findings. AJR Am J Roentgenol 2017;208:84-91.  Back to cited text no. 2
    
3.
Epstein S, Naji AF. Pulmonary artery aneurysm ith dissection after Blalock operation for tetralogy of Fallot. Am J Cardiol 1960;5:560-3.  Back to cited text no. 3
    
4.
Boubaker A, Payot M, Genton CY. Fatal rupture of an acquired aneurysm of the pulmonary artery: Rare complication after surgical palliation of tricuspid atresia. Pediatr Cardiol 1997;18:392-5.  Back to cited text no. 4
    
5.
Kumar RV, Roughneen PT, de Leval MR. Mycotic pulmonary artery aneurysm folloing pulmonary artery banding. Eur J Cardiothorac Surg 1994;8:665-6.  Back to cited text no. 5
    
6.
Knoles KA, Alvin M, Dunn E. Mycotic pulmonary artery aneurysm secondary to septic emboli. Case Reports 2017;2017.  Back to cited text no. 6
    
7.
Bartter T, Irin RS, Nash G. Aneurysms of the pulmonary arteries. Chest 1988;94:1065-75.  Back to cited text no. 7
    
8.
Kim HS, Oh YW, Noh HJ, Lee KY, Kang EY, Lee SY. Mycotic pulmonary artery aneurysm as an unusual complication of thoracic actinomycosis. Korean J Radiol 2004;5:68-71.  Back to cited text no. 8
    
9.
Kauffman SL, Lynfield J, Hennigar GR. Mycotic aneurysms of the intrapulmonary arteries. Circulation 1967;35:90-9.  Back to cited text no. 9
    
10.
Trambert JJ, Abubaker SJ, Kanner BJ. Giant mycotic pulmonary artery pseudoaneurysm treated by guide ire and coil embolization. J Vasc Interv Radiol 2014;25:1643-5.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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