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Int J Surg Case Rep
Int J Surg Case Rep
International Journal of Surgery Case Reports
2210-2612
Elsevier

S2210-2612(24)00961-1
10.1016/j.ijscr.2024.110180
110180
Case Report
A surgical odyssey: Conquering a titanic tricuspid vegetation reaching into the superior vena cava - A case report
Safdar Muhammad Nabeel nabeel.safdar2@aku.edu
⁎
Sohail Abdul Ahad
Iqbal Sara
Sami Shahid
Sharif Hasanat
Shahabuddin Syed
Section of Cardiothoracic Surgery, Department of Surgery, Aga Khan University Hospital, Karachi, Pakistan
⁎ Corresponding author. nabeel.safdar2@aku.edu
15 8 2024
10 2024
15 8 2024
123 11018011 7 2024
12 8 2024
14 8 2024
© 2024 Published by Elsevier Ltd on behalf of IJS Publishing Group Limited.
2024

https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Introduction

Right-sided infective endocarditis, particularly of the tricuspid valve, is rare and challenging to diagnose, often presenting with nonspecific symptoms, and associated with high mortality rates. This case underscores the complexities in managing such conditions and the importance of early diagnosis and multidisciplinary intervention.

Case presentation

This case study details the medical history of a 34-year-old woman who had a background of intravenous drug abuse. She subsequently developed a fungal tricuspid valve endocarditis, leading to the formation of vegetation that extended into the superior vena cava. The mass measured 15 × 3 cm upon surgical removal.

Discussion

Right-sided infective endocarditis is rare, comprising only 5–10 % of cases, with tricuspid valve endocarditis being even rarer. Damage to the endothelium facilitates bacterial attachment, especially in IV drug users, with Staphylococcus aureus being common. Fungal endocarditis is rare but deadly, with high mortality. Diagnosis relies on the modified Duke criteria, including microbiological and imaging evidence. Major complications affect both valvular and systemic areas. Treatment p1rimarily involves IV antibiotics, but surgery is needed for persistent infections or severe complications.

Conclusion

This case underscores the critical importance of early diagnosis and intervention in managing right-sided infective endocarditis, especially with a fungal pathology and in patients with complex medical histories.

Highlights

• A young female intravenous drug user presenting with fungal endocarditis.

• Right-sided tricuspid endocarditis is rare, hard to diagnose, and often fatal due to vague symptoms.

• Fungal tricuspid endocarditis, forming a massive mass extending into the Superior Vena Cava.

• Surgical resection of tricuspid vegetation and bicuspidization of the tricuspid valve.

Keywords

Infective endocarditis
Tricuspid valve
Intravenous drug use
Cardiac surgery
Case report
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pmc1 Introduction

Infective endocarditis (IE) is a condition affecting the inner lining of the heart known as the endocardium. Typically, the infection targets either the natural or artificial cardiac valves, or a cardiac device that has been implanted [1]. Infective endocarditis is a rare disease with an incidence rate of approximately 5 cases per 100,000 individuals annually [2]. The clinical manifestation of infective endocarditis exhibits a wide range of diversity and lacks specific characteristics [3]. Even with the progression of healthcare and guidelines, infective endocarditis remains a disease with poor outcomes [2]. In this intriguing case report, we explore the medical journey of a 34-year-old female whose history of drug abuse led to the development of a massive tricuspid vegetation, presenting an unprecedented challenge in the operating theater. This report also highlights the complex procedure necessitated by a delayed diagnosis and documents a massive vegetation, contributing to the limited literature on such cases.

This case report has been prepared in accordance with the SCARE criteria [4].

2 Case report

A 34-year-old female with a history of morbid obesity (weight: 130 kg, BMI: 41.5 kg/m2), depression, and intravenous (IV) drug abuse presented to our emergency department after leaving against medical advice (LAMA) from another hospital where she was being evaluated for a high-grade fever lasting four days, nausea, decreased appetite, and shortness of breath. At the previous hospital, she had been admitted to the intensive care unit (ICU) for investigation of the causes of her fever.

Her medical history included intravenous (IV) opioid abuse over the past five years, which had led to bilateral thigh cellulitis at injection sites four months ago. This condition required wound debridement and the insertion of a peripherally inserted central catheter (PICC) line for prolonged antibiotic therapy. Unfortunately, she subsequently used the PICC line for IV opioid abuse.

Diagnostic tests revealed a positive COVID-19 BioFire, elevated inflammatory and fungal markers, and a large mobile tricuspid valve vegetation (23 × 11 mm) on echocardiography. She was diagnosed with COVID-19 pneumonia, sepsis secondary to thigh wounds, infective endocarditis, and bicytopenia with low hemoglobin and platelet counts. Initial conservative management included broad-spectrum antibiotics and antifungals. Despite a surgical recommendation, the family refused, leading to her leaving against medical advice (LAMA) and subsequently presenting to our emergency room (ER).

Upon arrival at the ER, she was hemodynamically stable. Physical examination revealed multiple scars from previous injection sites and healed bilateral thigh wounds. She was admitted for further evaluation.

Initial investigations at our institute included a transthoracic echocardiogram and a computed tomography (CT) scan of the chest. The echocardiogram revealed an ejection fraction of 55–60 %, no segmental wall motion abnormalities, mildly reduced right ventricular (RV) systolic function, and RV pressure overload with a D-shaped left ventricular (LV) cavity during diastole. A positive McConnell sign suggested a pulmonary embolism. Additionally, there was a very large mobile mass on the tricuspid valve (7.2 × 1.7 cm), indicative of vegetation, with the possibility of a tumor, and a trace circumferential pericardial effusion. The CT scan of the chest showed no evidence of pulmonary embolism but identified filling defects in the superior vena cava (SVC) and right atrium, suggesting partial thrombosis. It also revealed consolidation with air bronchograms in the left lung apex and bilateral lower lobes, along with infiltrates in the bilateral upper lobes, indicating a pulmonary infection.

Laboratory investigations on arrival showed a hemoglobin level of 9.2 g/dL, a white blood cell count of 40.9 × 109/L (with neutrophils at 91.5 % and lymphocytes at 3.4 %), and platelets at 25 × 109/L. The C-reactive protein (CRP) level was 132 mg/L, galactomannan was 0.14 (negative), and Beta-D-glucan (BDG) was 500 (normal <80). Further workup showed negative results for HIV and ANA profiles. Additionally, initial blood cultures grew Candida albicans, and cultures from the PICC line grew Staphylococcus species, Candida albicans, and Pseudomonas aeruginosa.

On the third day of admission, the patient developed respiratory distress and type 1 respiratory failure, necessitating intubation and transfer to the ICU. Despite multidisciplinary management by hematology, infectious disease, cardiology, medical ICU, and cardiothoracic surgery teams, her condition deteriorated. Platelet counts only increased to 57 × 109/L, and hemoglobin dropped to 8.7 g/dL even after multiple transfusions. She was diagnosed with consumptive coagulopathy secondary to worsening sepsis and mechanical platelet destruction. After thorough counseling, the family consented to high-risk surgery. Preoperative lab investigations showed a WBC count of 28.0 × 109/L (Neutrophils: 89.9 %, Lymphocytes: 4.4 %), BUN at 41 mg/dL, creatinine at 1.9 mg/dL, normal electrolytes, prothrombin time (PT) of 13.4 s, INR of 1.3, activated partial thromboplastin time (APTT) of 25.5 s, procalcitonin at 2.44 ng/mL, CRP at 346 mg/L, and deranged liver function tests (LFTs).

The patient underwent excision of tricuspid valve vegetation and tricuspid valve repair. The procedure involved a midline sternotomy, pericardiotomy, and heparinization. Aortic cannulation was performed, and a palpable thrombus was noted in the Superior Vena Cava. Only a single-stage straight venous cannula was passed into the right atrial appendage, and cardiopulmonary bypass (CPB) was established, then another single stage straight venous cannula was passed in Inferior Vena Cava and full flows were achieved.

A cross-clamp was applied, and antegrade cardioplegia was administered to achieve diastolic arrest. The right atrial (RA) appendage cannula was clamped. The RA was opened after snugging the IVC, and massive flow was encountered from the SVC, managed with two cardiotomy suckers. Manual evacuation of the thrombus from the SVC was performed (Fig. 1), leading to further increased flow. Intraoperative findings included a large, thrombosed vegetation measuring 15 × 3 cm in the right atrium attached to the posterior leaflet of the tricuspid valve (Figs. 2 and 3).Fig. 1 Vegetation being carefully taken out from the right atrium.

Fig. 1

Fig. 2 Vegetation completely in right atrium.

Fig. 2

Fig. 3 Panoramic view of the excised mass.

Fig. 3

The right atrial appendage cannula was then advanced into the SVC, snugged, and declamped. Complete excision of the remaining vegetation was performed, and the posterior leaflet of the tricuspid valve was excised (Fig. 4). The area was thoroughly irrigated. Since the remaining leaflets were normal, bicuspidization of the anterior and septal leaflets was performed using 4/0 prolene pledgeted sutures (Fig. 5). A saline test showed reasonable coaptation of the leaflets with some tricuspid regurgitation. Finally, the RA was closed, de-aired, and the cross-clamp was removed. The patient was weaned off CPB smoothly without significant bleeding or increased chest drain output postoperatively.

Postoperatively, she required persistent high inotropic and ventilatory support. Despite initial improvement in platelet counts (114 × 109 on 1st Post Operative Day), her multi-organ dysfunction worsened due to septic shock, leading to hepatic and renal failure requiring dialysis. A limited postoperative echocardiogram revealed mild to moderate RV systolic dysfunction with free tricuspid regurgitation. Unfortunately, she passed away on the fourth postoperative day.

Cultures of the vegetation and tricuspid valve showed Candida albicans, while histopathology revealed fibrocollagenous tissue with acute and chronic inflammation and aggregates of fungal pseudohyphae and yeasts.Fig. 4 Excised posterior leaflet of the tricuspid valve.

Fig. 4

Fig. 5 Bicuspidization being done between septal and anterior leaflets with pledgeted prolene sutures.

Fig. 5

3 Discussion

Right-sided infective endocarditis stands as a rare gem within the realm of cardiac illnesses, making up only 5–10 % of all infective endocarditis cases [5]. In older literature, tricuspid valve endocarditis comprised only 2.5–3.1 % of all cases of infective endocarditis [6]. Right-sided infective endocarditis is often associated with intravenous drug use (IVDU), intracardiac devices, and central venous catheters. It is less common than left-sided infective endocarditis due to the lower prevalence of right-sided congenital anomalies, the unique properties of the right-sided endocardium, and the distinct hemodynamics experienced by the right-side endocardium [7].

The endothelium of a healthy heart can withstand frequent instances of bacteria entering the bloodstream due to everyday actions like chewing and brushing teeth [8]. However, following damage to the endothelium, the release of inflammatory cytokines and tissue factors along with increased fibronectin expression contributes to the creation of a platelet-fibrin complex, which facilitates bacterial attachment [9]. The persistent exposure of pathogen and particulate matter in a patient with frequent illicit drug use can damage and seed bacteria on a previously healthy native tricuspid valve [5]. This was a significant risk factor in our patient, who was not only an IV drug user but also had a PICC line in place that was used for illicit drug administration.

Various pathogens can cause endocarditis, as indicated in the literature. Among patients who use intravenous drugs, a prevalent offender is Staphylococcus aureus [10]. Fungal causes, as showcased in our case, are exceptionally uncommon, comprising only 3 % of all endocarditis cases. Their occurrence on the right side of the heart is even rarer [11]. Moreover, fungal endocarditis presents a formidable challenge with increased mortality rates as high as 54 % reported in one study [12].

Infective endocarditis has a varied and vague clinical presentation, making it challenging to diagnose. The condition is frequently overlooked or misdiagnosed, leading to delays in treatment and increased mortality rates. It is crucial to consider infective endocarditis in individuals with sepsis of unknown cause or fever along with relevant risk factors. Clinical examination reveals variable signs of disease, with fever present in approximately 90 % of cases and a cardiac murmur in about 85 %, being the most common [3]. Lastly, using well established criteria like the modified duke criteria can help in evaluating patients with a suspicion of infective endocarditis potentially leading to early intervention and better outcomes.

The modified Duke criteria diagnose infective endocarditis (IE) through major and minor criteria. A definite diagnosis requires two major criteria, one major with three minor, or five minor criteria. Major criteria include microbiological evidence, imaging findings of cardiac abnormalities, and direct surgical evidence. Minor criteria encompass predisposing factors, fever, vascular complications, immunological signs, microbiological evidence not meeting major criteria, and specific imaging abnormalities or new valvular regurgitation [13]. In our case, we identified a mass on echocardiography and obtained positive blood cultures that met the Duke criteria for diagnosing infective endocarditis.

The major complications of right-sided infective endocarditis encompass both valvular/local and nonvalvular/peripheral areas. Valvular issues include insufficiency (more commonly tricuspid regurgitation), stenosis, destruction, leaflet perforation, and annulus abscess formation. On the nonvalvular side, there are pulmonary complications (like embolism, infiltrates, and abscess), systemic embolism and infarcts, high-degree atrioventricular block, septal shunt-related embolism, septic shock, and multiorgan failure. These complications underline the wide-ranging impact of infective endocarditis, stressing the importance of thorough monitoring and management strategies to address both valvular and systemic ramifications effectively [7]. In our patient, the complications of right-sided infective endocarditis caused significant health deterioration, initially presenting with sepsis and eventually progressing to multiorgan failure. Furthermore, the challenges in obtaining consent for surgery at another hospital, followed by a transfer to our facility, led to an overall delay in management, contributing to her worsening condition.

IV antibiotics are the primary treatment for right-sided infective endocarditis affecting the tricuspid valve. Nevertheless, surgical intervention might be necessary in various circumstances.

Surgery is indicated in cases where microorganisms are challenging to eliminate (such as persistent fungi), bacteremia persists for over 7 days despite proper antimicrobial treatment (e.g., Staphylococcus aureus, Pseudomonas aeruginosa), tricuspid valve vegetations are large and persistent (>20 mm), recurrent pulmonary emboli occur with or without concurrent right heart failure, severe tricuspid regurgitation leads to right heart failure, or abscesses are present [14]. Our case had clear surgical indications due to massive vegetation and fungal pathology, coupled with deteriorating health despite medical management.

The goals of surgery involve eliminating infection and restoring the structure of the heart. Options for reconstruction include both repairing and replacing the valve, with no conclusive evidence favoring either a bioprosthetic or mechanical valve replacement [3]. In a retrospective study involving intravenous drug users, the primary factors associated with mortality were the length of vegetation exceeding 20 mm and the presence of fungal origins in right-sided infective endocarditis [7].

4 Conclusion

This report highlights a rare and life-threatening pathology with a very high mortality rate, underscoring the need for heightened awareness and early intervention. It raises important questions about whether current clinical guidelines should be revisited to incorporate more effective treatment modalities specifically targeting fungal right-sided endocarditis. Addressing these considerations could significantly improve patient outcomes and reduce mortality associated with this challenging condition. Although this case report describes a very rare and complex pathology and management, it inherently lacks sufficient evidence to support changes. However, it contributes to the existing literature on the poor outcomes of infective endocarditis, potentially prompting studies focused on different management strategies and outcomes.

Consent

Written informed consent was obtained from the patient for publication and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Ethical approval

Since this is a case report, it has received exemption by the Institutional Review Board at Aga Khan University, Karachi, Pakistan.

Funding

No funding was provided for this study.

Author contribution

Case report conception: Syed Shahabuddin.

Data collection: Abdul Ahad Sohail, Muhammad Nabeel Safdar.

Manuscript writing (First draft): Muhammad Nabeel Safdar.

Editing and critical review of the manuscript: Abdul Ahad Sohail, Sara Iqbal, Shahid Sami, Syed Shahabuddin.

Guarantor

Syed Shahabuddin.

Research registration number

N/A.

Conflict of interest statement

None.
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