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Background: Infective endocarditis (IE) is a rare disease with high mortality. Right-sided IE accounts for 5–10% of cases of IE. The tricuspid valve is most commonly affected, oppositely in coronary sinus (CS). The diagnoses, treatments and outcomes of CS vegetation has not been summarized yet. Case presentation: We present a 71-year-old man complained of cough and fever. Transthoracic echocardiography revealed the aneurysmal dilated CS with the band medium-echo mobile structure. A sinus venosus atrial septal defect has been detected. He had a persistent left superior vena cava which drained the right atrium via the aneurysmal dilated CS. Blood cultures were positive for Staphylococcus aureus. After intravenous antibiotic therapy, he had the symptom of dyspnea. The suspicious diagnosis is recurrent septic lung emboli which was confirmed by thoracic contrast enhanced computed tomography. The thoracotomy was performed to repair the atrial septum and remove the CS vegetation. Ten days later, the patient was discharged with only mild cough. Conclusion: Both positive blood cultures and echocardiography are major criteria in right-sided IE with CS vegetation. Current treatment options of CS vegetation include medical therapy and surgery. The surgical strategy for CS vegetation should be individualized, due to the controversial indications and optimum time of surgery. Most people have a good prognosis after proper treatment. Keywords: Coronary sinus, Infective endocarditis, Vegetation, Surgery, Echocardiography Background commonly affected, oppositely in coronary sinus Infective endocarditis (IE) is a rare disease with (CS) . Herein, we present the case of a prevalence ranging from 3 to 10 per 100,000 71-year-old man with right-sided IE and CS person-years . The average age of patients with vegetation. infective endocarditis are increasing from 40 shifting to 70 . Duetoits high mortality(20–25%) in the Case presentation past two decades, IE is now the third or fourth A 71-year-old previously healthy man complained of most common life-threatening infection syndrome cough and fever for 1 month. At first, he was evalu- [1, 3, 4]. According to its principal associated com- ated at his local hospital, where he was believed to plications in different clinical scenarios, IE could be have pneumonia. During hospitalization, he got sud- divided into several types, including the left-sided den chest pain and hemoptysis which were similar IE, right-sided IE, prosthetic valve IE, electronic de- as the symptoms of the pulmonary embolism. How- vices IE, and so on. Right-sided IE accounts for 5– ever, lower extremity doppler ultrasound didn’t 10% of cases of IE . Thetricuspid valveis most found any sign of thrombus. He presented to our hospital for a definite diagnosis. On day 1, his pulse * Correspondence: firstname.lastname@example.org Department of Ultrasound, Shengjing Hospital of China Medical University, Shenyang, China Full list of author information is available at the end of the article © The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Song et al. BMC Cardiovascular Disorders (2018) 18:111 Page 2 of 7 rate was 106 beats/minute, blood pressure was 134/ also positive. We believed these symptoms caused by 83 mmHg, and his temperature was 38.3 °C on recurrent septic lung emboli. Emergency thoracic examination. There were no murmurs on ausculta- contrast enhanced computed tomography was per- tion of the heart. Laboratory testing revealed a formed and revealed filling defects in the branches white blood cell count of 21,000/μL (neutrophils of the left lower pulmonary artery (Fig. 5a, b). Sinus 74.5%), hemoglobin of 11.5 g/dL, and platelets of venosus ASD has been confirmed (Fig. 6a, b). A per- 192,000/μL. Also, the C-reactive protein level is sistent left superior vena cava drained into the right 74 mg/L, and erythrocyte sedimentation rate is atrium through the aneurysmal dilated CS (Fig. 6c). 66 mm/h. The coagulation function is normal. The On day 8, thoracotomy was performed (Fig. 7a). A electrocardiogram showed sinus tachycardia without photograph of the gross specimen showed a netlike other abnormalities. On day 2, transthoracic echocar- vegetation which was removed from the CS (Fig. 7b). diography (TTE) revealed the aneurysmal dilated CS The vegetation was mixed with white and dark red. (diameter: 38 mm) with the band medium-echo mo- Histologic sectioning revealed that vegetation con- bile structure in the parasternal left ventricle tained a large number of necrotic material (Fig. 7c). long-axis view (Fig. 1a; Additional file 1: Movie 1). After surgery, his condition became stable. On day In the modified apical 4-chamber view, the band 18, the blood culture and other laboratory testing medium-echo mobile structure (40 × 12 mm) could normalized. The patient was discharged with only be observed in the aneurysmal dilated CS (Fig. 1b; mild cough. After he got home, he also had Additional file 2: Movie 2). A sinus venosus atrial two-week antibiotherapy in his local hospital. Two septal defect (ASD) with bi-directional shunt has months later, he came to the outpatient department been detected near the entrance of superior vena for follow-up. He was doing well without any com- cava in the right atrium (Fig. 2a,b,c). Part of severe plications. TTE only revealed the dilated CS and tricuspid regurgitation drained into the CS (Fig. 3). pericardial effusion. The persistent left superior vena cava has been re- vealed in the suprasternal long axis view of aortic Discussions and Conclusions arch (Fig. 4). Enlarged right heart, pericardial effu- Unlike the left-sided IE mainly occurred on the sion, dilated inferior vena cava may indicate dysfunc- aorta or mitral valve, right-sided IE could involve tional right heart. Moderate pulmonary artery the tricuspid valve, pulmonary valve, eustachian hypertension also has been revealed. On day 4, blood valve, interventricular septum, right ventricular free cultures were positive for Staphylococcus aureus wall, or CS. The right-sided IE with CS vegetation, which is methicillin sensitive. We highly suspect that just like our case, is extremely rare. So far, we have this is IE with CS vegetation. So, he got intravenous found 7 cases describing CS vegetation (Table 1) antibiotic therapy which lasted 2 weeks during [6–11]. There were 3 men and 4 women. Ages hospitalization. Cloxacillin is given by intravenous ranged from 23 years to 71 years; mean age 39.6 ± injection as 12 g/day in 4–6 doses. However, on day 19.8 years. Right-sided IE usually occurs in intraven- 6, the patient had symptoms of dyspnea and chest ous drug abusers or patients with human immuno- pain. We repeated the blood cultures which were deficiency virus [10, 11], cardiac device infection, Fig. 1 The aneurysmal dilated coronary sinus with the banded medium-echo mobile structure (yellow arrow). a in the parasternal left ventricle long-axis view. b In the modified apical 4-chamber view. CS: Coronary sinus; LA: Left atrium; LV: Left ventricle; PE: Pericardial effusion; RV: Right ventricle Song et al. BMC Cardiovascular Disorders (2018) 18:111 Page 3 of 7 Fig. 2 Sinus venosus atrial septal defect. a The size of echo drop is 22.3 mm. b, c bi-directional shunt has been detected between right atrium and left atrium. LA: Left atrium; LV: Left ventricle; RA: Right atrium; RV: Right ventricle central venous catheter, congenital heart disease, vegetation is the landmark lesion of IE. Vegetation in the and hemodialysis . These risk factors may cause CS has some characters: First, vegetation is usually iso- cardiac endothelial damage . The most common lated, may not affect other valves. Second, the CS always symptom of these patients is fever. Due to the pos- dilated. Third, vegetation in the CS is usually big (length > sibility of abscission of vegetation, right-sided IE 10 mm), shaped like a tubule mass. Our case is the only with CS vegetation could present with a complica- netlike one. Most of CS vegetations are mobile. Echocardi- tion, particularly septic shock or pulmonary embol- ography could detect of the associated cardiovascular ism (hemoptysis). anomalies, including coronary artery-CS fistula [6, 7, 9], The first modality for diagnosis is TTE (71%) and ASD, and valvular regurgitation. Echocardiography also transesophageal echocardiography (29%). Echocardi- could provide information about severity of the valve le- ography is crucial to diagnosis. TTE is a first line sion, and assess the left/right ventricular function . imaging study in the diagnosis of IE at present . Blood culture is crucial as a major criterion for the In suspected IE, TTE has a moderate sensitivity diagnosis of right-sided IE. In these seven patients, (75%) and high specificity (> 90%) in detecting IE about 71% (5/7) of blood cultures were positive. with vegetation . In patients with an equivocal Pathogenic bacteria included Staphylococcus aureus or negative TTE, but high clinical likelihood of (2), Streptococcus (2), and Acinetobacter baumanii infective endocarditis, transesophageal echocardiography (1). Similar results have been found by previous is necessary due to the higher sensitivity (> 90%). Each of studies which revealed that Staphylococcus aureus three positive echocardiographic findings which include was the most common cause of right-sided IE [15, vegetation, cardiac abscess, and new valvular regurgitation 16]. Staphylococcus aureus infection has been shown could provide sufficient evidence of IE . In particular, to be an independent predictor of mortality from IE Fig. 3 Severe tricuspid regurgitation has been revealed, part of Fig. 4 The persistent left superior vena cava has been revealed in which drained into the coronary sinus. CS: Coronary sinus; RA: Right the suprasternal long axis view of aortic arch. PLSVC: Persistent left atrium; RV: Right ventricle superior vena cava Song et al. BMC Cardiovascular Disorders (2018) 18:111 Page 4 of 7 Fig. 5 Emergency thoracic contrast enhanced computed tomography revealed pulmonary embolism. a, b filling defects in the branches of the left lower pulmonary artery (yellow arrow) . Doctor must pay special attention to this kind Treatment for at least 4–6 weeks is usually necessary. of microorganism because Staphylococcus aureus has Although undertaken in 40–50% of patients with IE [3, a higher mortality (51%) than others (31%) . The 21], the necessity and indications of surgery main con- high mortality may due to its complicated large veg- troversial. According to previous studies and guidelines, etations, invasive valve damage, and embolic symp- indications for surgery of IE have been summarized [22– tom . The negative result of blood culture has a 24] (Table 2). The purpose of surgery is to eradicate the incidence of 2.5–31%, which could delay diagnosis infection and achieve hemodynamic correction. The rea- and the initiation of treatment . son for surgery in our case is as follow: (1) Patients may Current treatment options of IE include medical ther- have recurrent septic pulmonary emboli on his sixth day apy and surgery. Most of the patients with CS vegetation in hospital. (2) Patients with severe tricuspid regurgita- (86%) received antibiotic therapy. Medical therapy is the tion and dysfunctional right heart. (3) The size of vege- primary treatment strategy . On an empirical basis, tation larger than 10 mm. (4) The patient has a risk of antibiotics should be started as soon as blood cultures paradoxical embolism due to associating ASD with have been acquired, but doctors could also await result bi-directional shunt. Cerebrovascular complications, of blood culture if the condition of patient is stable . causing by paradoxical embolism, could decease the Fig. 6 Emergency thoracic contrast enhanced computed tomography revealed cardiovascular anomalies. a Contrast agent slightly appeared in left atrium (yellow arrow) when the right heart was enhancing. b Interruption was visible between right atrium and left atrium. c A persistent left superior vena cava drained into the right atrium through the aneurysmal dilated coronary sinus Song et al. BMC Cardiovascular Disorders (2018) 18:111 Page 5 of 7 Fig. 7 a Thoracotomy was performed to repair the atrial septum and remove the coronary sinus vegetation. b Photograph of the gross specimen showed a netlike vegetation which was removed from the coronary sinus. The vegetation was mixed with white and dark red. c Histologic sectioning revealed that vegetation contained a large number of necrotic material and neutrophils quality of life in the long term. The optimum time of patients have an uneventful recovery. Only one pa- surgery remains indistinct. Previous study revealed tient died due to multiple organ failure after her that early surgery should be considered early if surgery. Staphylococcus aureus is suspected . Half of sur- Both positive blood cultures and echocardiography are geries were performed in the acute phase, the other major criteria in right-sided IE with CS vegetation. half in the convalescent phase . Current treatment options of right-sided IE include Prognosis of right-sided IE is usually well. Previous medical therapy and surgery. The surgical strategy for study revealed the mortality of right-sided IE is 12% right-sided IE patients with CS vegetation should be in- in-hospital patients , and 0–7.3% for surgical pa- dividualized, due to the controversial indications and tients [27, 28]. In these seven patients with CS vege- optimum time of surgery. Most people have a good tation, the in-hospital mortality is 14%. Most of the prognosis after proper treatment. Table 1 Summary of literature involving right-sided IE with CS vegetation No. First Author Year Sex, age Symptoms First Length × Associated Blood culture Treatments Outcomes modality width of cardiovascular for diagnosis vegetation (mm) anomalies a a 1 Kasravi  2004 M, 31y Fever, chills, TTE > 27 ×6 CACSF, Positive for Antibiotic Alive nausea, vomiting, vegetation Staphylococcus myalgias, extends to RA aureus neck stiffness 2 Gill  2005 M, 37y Fever, weight loss TEE 14 × 7 CACSF Positive for Antibiotic Alive Streptococcus 3 Kwan  2014 F, 23y Fever TTE 14 × 2 Vegetation Positive for Antibiotic Alive extends to RA Acinetobacter baumanii 4 Takashima  2016 F, 64y Fever, fatigue, TTE 17 × 9 CACSF, Negative Surgery Died loss of appetite, vegetations on septic shock the mitral and aortic valves with moderate regurgitation, heart failure 5 Kumar  2017 F, 23y Septic shock TEE 30 × 5 Vegetation on the / Antibiotic Alive Eustachian valve in the RA a a 6 Theodoropoulos  2017 F, 28y Fever, sweat TTE 15 ×8 Tricuspid valves Positive for Antibiotic Alive malaise, hemoptysis, with moderate Streptococcus dyspnea regurgitation 7 Our case 2017 M, 71y Fever, cough, TTE 40 × 12 ASD, PLSVC, Positive for Antibiotic, surgery Alive chest pain, tricuspid valves Staphylococcus hemoptysis, dyspnea with severe aureus regurgitation ASD Atrial septal defect, CACSF Coronary artery-coronary sinus fistula, PLSVC Persistent left superior vena cava, RA Right atrium, TEE Transesophageal echocardiography, TTE Transthoracic echocardiography : measured from the figures in the literature Song et al. BMC Cardiovascular Disorders (2018) 18:111 Page 6 of 7 Table 2 Indications for surgery of IE according to the previous Competing interests The authors declare that they have no competing interests. studies and guidelines [22–24] 1. Patients with persistent infection who do not respond to antibiotic therapy beyond 2 weeks, except for specific pathogens that aggressive Publisher’sNote treatment should be considered early in the course of the disease (e.g. Springer Nature remains neutral with regard to jurisdictional claims in Staphylococcus aureus, Gram negative fungi); Perivalvular extension: published maps and institutional affiliations. abscesses, fistulas. Author details 2. Patients with recurrent septic pulmonary emboli, confirmed by Department of Ultrasound, Shengjing Hospital of China Medical University, computed tomography pulmonary angiogram. Shenyang, China. Department of Pathology, Shengjing Hospital of China 3. Patients with massive or worsening tricuspid regurgitation (> 2+/4+) 3 Medical University, Shenyang, China. Department of Radiology, Shengjing contributing to deteriorating right (and subsequently impending left) 4 Hospital of China Medical University, Shenyang, China. Department of ventricular heart failure; evaluated by echocardiography. Cardiac Surgery, Shengjing Hospital of China Medical University, Shenyang, China. 4. Patients in septic shock and documented right-sided IE (indication for emergency operation). Received: 27 December 2017 Accepted: 23 May 2018 5. When the size of a vegetation increases or persists in spite of antibiotic management at > 10 mm. 6. New-onset acute or worsening renal and/or hepatic failure. References 1. Baddour LM, Wilson WR, Bayer AS, Fowler VG Jr, Tleyjeh IM, Rybak MJ, Barsic 7. Patients with right-sided IE who develop a secondary (right- or left- B, Lockhart PB, Gewitz MH, Levison ME, et al. 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BMC Cardiovascular Disorders – Springer Journals
Published: Jun 4, 2018
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