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Abstract
A man in his early 30s presenting with chest pain was admitted for the management of acute pericarditis and evaluation of a subcarinal mass incidentally noted on chest imaging. Shortly after admission, he developed cardiac tamponade. Emergent pericardiocentesis revealed purulent pericardial fluid with polymicrobial anaerobic bacteria, raising concern for gastrointestinal source and broad intravenous antibiotics were given. The pericardial fluid reaccumulated despite an indwelling pericardial drain and intrapericardial fibrinolytic therapy, necessitating a surgical pericardial window. Concurrent fluoroscopic oesophagram demonstrated oesophageal perforation with fistulous connection to the subcarinal mass and mediastinal drain, suggestive of oesophagopericardial fistula. Pathology from biopsy of the subcarinal mass returned with focal large necrotising granulomas consistent with histoplasmosis. Antifungal treatment was initiated, and the patient was eventually discharged home with nasogastric feeding tube and oral antibiotics and antifungals. This is the first reported case of polymicrobial pericarditis secondary to acquired oesophagopericardial fistula likely induced by mediastinal histoplasma lymphadenitis.
- Interventional cardiology
- Infectious diseases
- Adult intensive care
- Pathology
- Cardiothoracic surgery
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Background
Purulent pericarditis is rare in the antibiotic era but requires aggressive investigation and management once diagnosed.1 The presence of anaerobic, polymicrobial bacteria in the pericardial fluid should raise suspicion for gastrointestinal, orofacial and/or dental sources of seeding.2 Acquired oesophagopericardial fistula is an extremely rare aetiology and is associated with high mortality secondary to sepsis, haemorrhage or cardiac tamponade.3 Management with empirical intravenous antibiotics and pericardiocentesis should be urgent, and intrapericardial thrombolysis and/or subxiphoid pericardiostomy may be necessary due to high rates of fluid reaccumulation and loculation in purulent pericarditis.4
Case presentation
A man in his 30s with a history of cocaine use and no other known medical history presented to the emergency department with the acute onset of crushing substernal chest pain. The chest pain was worse when lying back and improved with leaning forward. He denied having any recent viral illnesses or symptoms of an upper respiratory tract infection. Review of systems was also negative for shortness of breath or constitutional symptoms, including fevers or chills, night sweats or unintentional weight loss. His last reported cocaine use was the night prior to admission. He denied having any medical problems, including diabetes mellitus or hyperlipidemia. He did not use tobacco. He had no premature family history of cardiovascular diseases. On physical examination, the patient was afebrile, tachycardic with a heart rate of 130 beats per minute and hypertensive to 180/110 mm Hg with normal oxygen saturation on room air. The examination was notable for uncomfortable appearance, regular tachycardia with no murmurs or rubs, no jugular venous distension, no lower extremity swelling, no chest wall tenderness and normal work of breathing on room air with clear lung fields. Serial ECG demonstrated sinus tachycardia with evolving diffuse ST segment elevation and PR segment depression (figure 1). Serial high-sensitivity troponin measurements remained within normal limits. B-type natriuretic peptide was also normal. CT angiography of the chest was negative for aortic dissection and pulmonary embolism but did note a small pericardial effusion and a 4.9×3.3 × 4.4 cm soft-tissue mass in the posterior mediastinum concerning a subcarinal mass (figure 2). Echocardiogram with Doppler demonstrated a left ventricular ejection fraction of 75%, a small circumferential pericardial effusion without echocardiographic evidence of tamponade, echogenic material in the pericardial space and no pericardial bounce. Cardiology was consulted and empirical treatment for acute pericarditis with colchicine and high-dose ibuprofen was initiated. The patient was admitted to the medicine service by which time his chest pain as well as tachycardia had improved. Further evaluation of the posterior mediastinal mass with positron emission tomography-computed tomography scan and biopsy was planned.
ECG on presentation demonstrating sinus tachycardia with diffuse ST segment elevation and PR segment depression consistent with acute pericarditis.
CT angiography of the chest in axial plane (panel A) and sagittal plane (panel B) demonstrating 4.9×3.3×4.4 cm soft-tissue mass in the posterior mediastinum concerning a subcarinal mass.
On the second day of admission, the patient complained of acute generalised malaise and was found to be tachycardic with a heart rate of 130 beats per minute and undetectable blood pressure using an automatic cuff. Physical exam was notable for diaphoresis, tachypnea and cool extremities with difficult-to-palpate peripheral pulses. Intravenous fluids, vancomycin and cefepime were initiated. ECG again demonstrated sinus tachycardia and diffuse ST elevation, however, now with electrical alternans. Point-of-care ultrasound revealed a large pericardial effusion. The cardiac intensive care unit and cath lab team were consulted for concern about cardiac tamponade. Within 30 min, the patient was requiring vasopressor support with norepinephrine, vasopressin and epinephrine. His blood pressure remained undetectable, and assessment for pulsus paradoxus was unable to be performed. He was transported to the cardiac catheterisation lab for emergent pericardiocentesis, and 850 mm of grossly purulent fluid was drained from the pericardial cavity with subsequent resolution of the patient’s haemodynamic instability. An indwelling drainage catheter was left in place, and broad-spectrum intravenous antibiotics were continued. Pericardial fluid cultures were positive for beta-haemolytic Streptococci Group C, Fusobacterium gonidiaformans, Eikenella corrodens, Streptococcus salivarius and Streptococcus mitis, raising concern for a gastrointestinal source of purulent pericarditis given the anaerobic, polymicrobial nature of the effusion. Blood cultures remained negative.
The pericardial effusion later reaccumulated (online supplemental video 1), despite the indwelling pericardial drain, with patient again developing haemodynamic instability, necessitating intrapericardial fibrinolytic therapy, followed by a surgical pericardial window. Concurrent biopsies of pericardium and subcarinal mass as well as oesophagogastroduodenoscopy (EGD) were performed. EGD was notable for a 1 cm oesophageal mucosal disruption; therefore, antifungal therapy with fluconazole was initiated, and a nasogastric feeding tube was inserted to allow healing of the oesophageal perforation. Further evaluation with fluoroscopic oesophagram demonstrated a 7 mm oesophageal perforation along the right side of the midoesophagus with leakage of contrast into the subcarinal mediastinum extending into the mediastinal drain, raising suspicion for an oesophagopericardial fistula (figure 3). Pathology from pericardial biopsy was consistent with fibrinous pericarditis. Pathology from the subcarinal mass biopsy returned with fragments of soft tissue/lymphoid tissue with focal large necrotising granulomas (figure 4). Grocott’s methenamine silver stain showed focal rare yeast forms (figure 4), while acid-fast bacilli (AFB) stain was negative. The subcarinal mass pathology results raised concern for histoplasmosis, which is endemic to the region; therefore, empirical treatment with itraconazole was initiated and fluconazole was discontinued. Otherwise, fungal cultures and AFB cultures from pericardial fluid remained negative. QuantiFERON Gold, serum cryptococcus antigen, serum coccidioides antibody, urine histoplasma antigen and urine blastomyces antigen were also negative.
Supplementary video
Fluoroscopic oesophagram demonstrating a 7 mm oesophageal perforation along the right side of the midoesophagus (panel A) with leakage of contrast into the subcarinal mediastinum extending into the mediastinal drain (panel B).
Pathology slides from the biopsy of the subcarinal mass demonstrating focal large necrotising granulomas in low-powered, medium-powered and high-powered fields (panels A, B and C, respectively) and focal rare yeast forms on Grocott’s methenamine silver stain (panel D).
The pericardial drain was removed 2 days following pericardial window, and repeat echocardiogram did not demonstrate residual effusion. Repeat EGD with intraoperative oesophagram demonstrated a persistent leak from the previously noted oesophageal disruption, so nasogastric feeding tube was maintained. The patient was discharged to his home with nasogastric feeding tube, 14-day course of oral amoxicillin–clavulanate and a course of itraconazole with duration to be determined at outpatient follow-up.
Outcome and follow-up
Repeat oesophagram 1 month after discharge did not demonstrate evidence of contrast leak, so the nasogastric feeding tube was removed, and the patient has tolerated advancement of diet by mouth. Itraconazole therapy is ongoing for a planned 6 month course.
Discussion
This case report describes a rare clinical presentation of a polymicrobial pericardial effusion leading to cardiac tamponade, caused by an oesophagopericardial fistula likely related to mediastinal histoplasma lymphadenitis. To our knowledge, this is the first case report of this nature described in the literature.
Purulent pericarditis is an uncommon pathology in the antibiotic era. A retrospective review of cases of purulent pericarditis in a Spanish hospital between 1972 and 1991 found only 33 cases out of a cumulative inpatient population of 593 600.1 The most commonly identified infectious sources were empyema or pneumonia.1 However, another study noted that a primary infectious source was only found in 22% of purulent pericarditis cases in the antibiotic era.5 Predisposing factors include pre-existing pericardial disease, immunosuppression, thoracic surgery, chest trauma and alcohol abuse.4 The most common pathogens implicated in purulent pericarditis include staphylococci, pneumococci and streptococci.1 In patients with the aforementioned predisposing factors, Staphylococcus aureus and fungi are more common.6 Purulent pericarditis with anaerobic organisms has also been reported in patients with mediastinitis following oesophageal perforation and in patients with orofacial and dental infections.2 Mechanisms by which anaerobic bacteria may seed the pericardial space include haematogenous spread, penetrating or iatrogenic spread or contiguous spread from a thoracic, cardiac or subdiaphragmatic source of infection.7 In our patient’s case, purulent pericarditis occurred via contiguous spread of anaerobic bacteria from his oesophageal perforation with fistulous connection to the pericardium.
Acquired oesophagopericardial fistula is rare and associated with particularly high mortality secondary to sepsis, haemorrhage and cardiac tamponade. A literature review noted only 60 cases of acquired oesophagopericardial fistula with a mortality rate of 83%.3 Most causes of oesophagopericardial fistula were benign oesophageal pathologies, such as oesophageal ulcer, reflux oesophagitis, foreign object ingestion, iatrogenic injury, tuberculosis, oesophageal diverticula, caustic substance ingestion, Barrett’s oesophagus and achalasia.3 Oesophageal malignancy was noted in 23% of cases.3 However, our patient did not have evidence of benign or malignant oesophageal disease on EGD nor a recent history of penetrating foreign objects, toxic ingestion or iatrogenic injury. Thus, the subcarinal mass suspected to be histoplasmosis infection must be considered as a possible aetiology. This is supported by fluoroscopic oesophagram demonstrating leakage from the oesophageal perforation into the subcarinal mediastinum that then extends into the mediastinal space. Cases of oesophageal fistula in association with histoplasmosis have been reported in the literature, including a case of fistulous connection between the oesophagus and a mediastinal mass identified as histoplasmosis capsulatum, as well as several cases of bronchoesophageal fistula resulting from primary mediastinal inflammation from histoplasmosis.8 9 Therefore, our patient’s subcarinal mass attributed to histoplasmosis likely induced mediastinal inflammation that resulted in fistulous connection between the oesophagus and pericardium, leading to subsequent purulent pericarditis.
Purulent pericarditis must be aggressively managed even in the absence of cardiac tamponade with urgent pericardial drainage and empirical intravenous antibiotic therapy.4 Bacterial, fungal and tuberculous studies should also be sent from pericardial fluid and blood cultures should be drawn.4 In addition to pericardiocentesis, drainage with intrapericardial thrombolysis or subxiphoid pericardiostomy with rinsing of pericardial space may need to be considered, given the high frequency of fluid reaccumulation and loculation in purulent pericarditis.10–12 In cases of thick and loculated effusion, dense adhesions, recurrent tamponade and/or progression to constriction, pericardiectomy may be necessary.4 In our case, the patient had initial resolution of cardiac tamponade with pericardiocentesis; however, he soon after had reaccumulation of pericardial fluid despite the presence of indwelling pericardial drain and required intrapericardial fibrinolytic therapy and surgical pericardial window. Fortunately, the patient has not required a pericardiectomy.
Learning points
Presence of anaerobic, polymicrobial bacteria in purulent pericardial fluid should raise suspicion for gastrointestinal, orofacial and/or dental sources. Seeding of the pericardium with these bacteria occurs through haematogenous spread, penetrating or iatrogenic spread or contiguous spread, and the underlying mechanism may require treatment for source control.
Acquired oesophagopericardial fistula is rare and associated with particularly high mortality from sepsis, haemorrhage and/or cardiac tamponade. The most commonly reported aetiologies include benign and malignant oesophageal disease. When history or endoscopic examination does not reveal primary oesophageal disease or oesophageal trauma/iatrogenic injury, an index of suspicion should be maintained for mediastinal inflammation as a cause of fistulous connection.
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References
Supplementary materials
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Footnotes
Contributors The following authors were responsible for drafting of the text, sourcing and editing of clinical images, investigation results, drawing original diagrams and algorithms, and critical revision for important intellectual content: AS, NB, LD and RS. AS was a resident involved in the patient’s care, performed literature review, drafted the manuscript and images and served as corresponding author. NB was a cardiology fellow involved in the patient’s care and reviewed the manuscript and images. LD was an interventional cardiology attending involved in the patient’s care and reviewed the manuscript and images. RS was an interventional cardiology attending involved in the patient’s care and reviewed the manuscript and images. RS. The guarantor is RS. The following authors gave final approval of the manuscript: AS, NB, LD and RS.
Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Case reports provide a valuable learning resource for the scientific community and can indicate areas of interest for future research. They should not be used in isolation to guide treatment choices or public health policy.
Competing interests None declared.
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