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Rare Presentation of Endocarditis and Mycotic Brain Aneurysm
Journal Pre-proof Rare Presentation of Endocarditis and Mycotic Brain Aneurysm Ziv Beckerman, MD, Luis E. Martínez-Bravo, Gregory Johnson, Byron Holt, Charles D. Fraser, MD PII:
S0003-4975(19)31173-7
DOI:
https://doi.org/10.1016/j.athoracsur.2019.06.073
Reference:
ATS 32896
To appear in:
The Annals of Thoracic Surgery
Received Date: 29 March 2019 Revised Date:
14 June 2019
Accepted Date: 20 June 2019
Please cite this article as: Beckerman Z, Martínez-Bravo LE, Johnson G, Holt B, Fraser CD, Rare Presentation of Endocarditis and Mycotic Brain Aneurysm, The Annals of Thoracic Surgery (2019), doi: https://doi.org/10.1016/j.athoracsur.2019.06.073. This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2019 by The Society of Thoracic Surgeons
1
Rare Presentation of Endocarditis and Mycotic Brain Aneurysm Running Head: Endocarditis and Mycotic Brain Aneurysm
AUTHORS: Ziv Beckerman, MD1, Luis E. Martínez-Bravo3,1, Gregory Johnson2, Byron Holt2, and Charles D. Fraser, MD.
AFFILIATION: 1Division of Pediatric and Congenital Cardiothoracic Surgery, Department of Surgery and Perioperative Care and 2Department of Pediatrics, University of Texas Dell Medical School, Dell Children’s Medical Center, Austin, Texas; and 3Tecnologico de Monterrey, Escuela de Medicina y Ciencias de la Salud, Monterrey, Mexico.
Disclosures: Authors have nothing to disclose with regard to commercial support
WORD COUNT: 1488
CORRESPONDING AUTHOR: Ziv Beckerman, MD Dell Children’s Medical Center, Center for Pediatric and Congenital Heart Disease, 4900 Mueller Blvd, Suite 3S.003, Austin, TX 78723 (zbeckerman@ austin.utexas.edu)
2
Abstract Bartonella endocarditis can be a very elusive diagnosis. The clinical manifestations can vary and, at times, include multiorgan involvement. This case report describes two patients presenting with multiorgan failure, cerebral mycotic aneurysms and valvular endocarditis secondary to Bartonella infection. The complex diagnosis, decision making, and surgical management are described.
3
This report describes two patients admitted to our institution one month apart of each other. Both had a very similar clinical presentation. Their respective case reports are described below, followed by a discussion.
Case Reports Case 1 The first patient is a 21-year-old male with a history of a bicuspid aortic valve. He had previously undergone balloon aortic valvuloplasty at the age of 9 years. He presented to our emergency department with high grade fever (39°C), acute renal failure, proteinuria, anemia and elevated Brain Natriuretic Peptide (BNP) (1453 pg/ml). His pertinent history included a 1-year history of night sweats, 2-months history of dry cough and 1-month history of bilateral ankle leg swelling. Transthoracic echocardiography revealed a thickened and dysplastic aortic valve, moderate to severe aortic insufficiency, mild aortic valve stenosis and moderately reduced left ventricular systolic function. No vegetations were seen. Concurrent work-up and treatment of both heart failure (with diuresis, inotrope infusion, and red blood cell infusion), renal failure, and suspected endocarditis were initiated. Broad spectrum empiric antibiotic coverage was started. His endocarditis work-up revealed Bartonella henselae on blood DNA testing. On a more focused anamnesis, the patient did report exposure to cats a few months previously. All other cultures came back negative. In addition, he underwent cerebral and cardiac Magnetic Resonance Imaging (MRI). His cerebral MRI demonstrated a small mycotic aneurysm, 2.5x2mm in the right middle cerebral artery (M3) distribution (Figure 1.). His cardiac MRI revealed a thickened bicuspid aortic valve with mild stenosis and severe regurgitation. Several multidisciplinary
4
discussions took place and the decision was made to address his mycotic brain aneurysm prior to surgical repair of his heart. Accordingly, he underwent cerebral angiography (Figure 2.) and following that, MRI-guided stereotactic right frontotemporal craniotomy and clip ligation of a mycotic aneurysm. Four weeks following his neurosurgery, he underwent aortic root replacement with a 24 mm aortic homograft valved conduit and closure of a patent foramen ovale via median sternotomy on cardiopulmonary bypass support. The operative findings were notable for a degenerated and necrotic aortic valve and aortic root abscess extending into the annulus of the right coronary artery cusp. Cross-clamp time was 174 minutes. Cardiopulmonary bypass time was 250 minutes. His postoperative recovery was uneventful, and he was discharged home 8 days after surgery. He completed 6 weeks of intravenous antibiotic treatment and continued doxycycline oral treatment for life. Case 2 The second patient is a 20-year-old male with a medical history of Noonan syndrome, von Willebrand disease, and asthma. He underwent pulmonary valve balloon valvuloplasty when he was 1 year of age. He presented with new onset symptoms of chest pain and shortness of breath. He was also found to have renal failure, severe proteinuria, BNP >5800 pg/ml, and anemia. His chest X-ray demonstrated pulmonary edema. Echocardiography demonstrated the mitral valve to be thickened with mild insufficiency and severe stenosis (mean pressure gradient of 1718mmHg), good biventricular function, and no echocardiographic evidence of vegetations. The infectious disease workup was positive for Bartonella vinsonii with blood DNA testing. Cerebral MRI demonstrated a 1.3 cm x 1 cm partially thrombosed left frontal middle cerebral artery mycotic aneurysm. Accordingly, he underwent a cerebral catheterization followed by a CTguided stereotactic left frontoparietal craniotomy for resection and ligation of the aneurysm. We
5
waited 4 weeks for him to recover from his neurosurgical intervention and proceeded with the cardiac repair. He underwent mitral valve replacement with a mechanical 29mm prosthesis. Operative findings were remarkable for congested lungs and engorged lymphatics. The left atrial wall was extremely thickened, the mitral valve itself was severely diseased with a miniscule orifice of 8-10 mm and thickening of the leaflets. There was obvious infection involving the anterior and posterior mitral valve leaflets. Cardiopulmonary bypass time was 171 minutes. Aortic cross-clamp time was 143 minutes. He was extubated on postoperative day-1 and did very well following that. He continued intravenous antibiotic treatment per the recommended protocol and continues oral doxycycline. His postoperative course was notable for a chylus pericardial effusion requiring drainage. Comment These two cases describe a unique presentation of Bartonella endocarditis with concurrent cardiac valvular disease and mycotic brain aneurysms. The initial presentation of both patients with heart failure, renal failure and stigmata of chronic illness while having an underlying cardiac pathology should alert the clinician for the possibility of an atypical endocarditis. The diagnosis of Bartonella endocarditis can be very elusive. Even though both patients had no evidence of vegetations by echocardiography, we elected to pursue all avenues to diagnose/rule out endocarditis. PCR-based diagnosis of Bartonella endocarditis and/or of other pathogens from blood has not been the standard recommended pathway for diagnosis of endocarditis neither in the adult or pediatric guidelines [1, 2]. That said, if we had not performed that assay, we would have been unlikely to identify the causative pathogen and would probably have not performed brain imaging. In that case, we would have potentially exposed those patients for neurological
6
complications secondary to their brain aneurysms during cardiac surgery. Additionally, neither of the patients would have received appropriate antibiotic coverage prior to their surgery. With regards to conduit/valve choices, undertaking an elaborate discussion with the patient and his family is paramount, as various surgical options, valves, and conduits exist. The first patient chose not to undergo a Ross procedure, and the second, chose a mechanical prosthesis after discussing all available surgical solutions (including a bioprosthesis with a potential for a future valve-in-valve option). The management of these patients is complex and requires a multidisciplinary approach to appropriately coordinate their care. Routine brain imaging is part of our workup for endocarditis, despite it being an area of controversy [3]. After diagnosing the brain aneurysms, we elected to address the cerebral issues first due to the concern of suffering a cerebral hemorrhage during open-heart surgery. Should an MRI of the brain be obtained in all infective endocarditis patients (or maybe only those with left-heart involvement) because cerebral emboli are so commonplace? No current consensus exists. Additionally, we had the concern of implanting a prosthetic cardiac valve while potentially having an infectious source elsewhere in the body. The decision to delay cardiac surgery in the setting of heart failure and acute valvular pathology can be questioned. Operating on the brain first results in a significant delay in the cardiac surgical care. The wait-time between the neuro- and cardiac surgeries, should be discussed by a multidisciplinary team, taking into consideration the pathogen’s behavior, the neurosurgical findings, the cardiac manifestations, and the clinical stability of the patient. These patients should remain in the intensive care unit as they can decompensate unexpectedly. If the patients are
7
deemed stable enough to undergo neurosurgical intervention first, it may be in their overall benefit for the long run. Additionally, addressing the cerebral issues first does allow for a longer course of antibiotic therapy prior to cardiac surgery. To conclude, (1) We advocate for extensive endocarditis evaluation including blood DNA testing whenever clinical suspicion exists even in the absence vegetations on echocardiography, (2) Diagnosis of endocarditis warrants cerebral imaging in our practice, (3) When clinically appropriate, the neurological findings should be addressed prior to performing a cardiac operation, and (4) A multidisciplinary approach is essential for caring of this complex patient group.
8
References 1. Baltimore RS, Gewitz M, Baddour LM, Beerman LB, Jackson MA, Lokhart PB, et al. Infective endocarditis in childhood: 2015 update. Circulation. 2015;132:1487–515. 2. Baddour L.M., Wilson W.R., Bayer A.S., Fowler V.G., Tleyieh I.M., Rybak M.J. Infective endocarditis in adults: diagnosis, antimicrobial therapy, and management of complications: a scientific statement for healthcare professionals from the American Heart Association. Circulation. 2015;132:1435–1486. 3. Champey J, Pavese P, Bouvaist H, Kastler A, Krainik A, Francois P. Value of brain MRI in infective endocarditis: a narrative literature review. Eur J Clin Microbiol Infect Dis. 2016;35:159–168.
9
Figure Legends Figure 1. Cerebral MRI demonstrating a cerebral aneurysm with hemorrhagic involvement. Figure 2. Cerebral angiography, a mycotic cerebral aneurysm in the right middle cerebral artery distribution.
S0003-4975(19)31173-7
DOI:
https://doi.org/10.1016/j.athoracsur.2019.06.073
Reference:
ATS 32896
To appear in:
The Annals of Thoracic Surgery
Received Date: 29 March 2019 Revised Date:
14 June 2019
Accepted Date: 20 June 2019
Please cite this article as: Beckerman Z, Martínez-Bravo LE, Johnson G, Holt B, Fraser CD, Rare Presentation of Endocarditis and Mycotic Brain Aneurysm, The Annals of Thoracic Surgery (2019), doi: https://doi.org/10.1016/j.athoracsur.2019.06.073. This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2019 by The Society of Thoracic Surgeons
1
Rare Presentation of Endocarditis and Mycotic Brain Aneurysm Running Head: Endocarditis and Mycotic Brain Aneurysm
AUTHORS: Ziv Beckerman, MD1, Luis E. Martínez-Bravo3,1, Gregory Johnson2, Byron Holt2, and Charles D. Fraser, MD.
AFFILIATION: 1Division of Pediatric and Congenital Cardiothoracic Surgery, Department of Surgery and Perioperative Care and 2Department of Pediatrics, University of Texas Dell Medical School, Dell Children’s Medical Center, Austin, Texas; and 3Tecnologico de Monterrey, Escuela de Medicina y Ciencias de la Salud, Monterrey, Mexico.
Disclosures: Authors have nothing to disclose with regard to commercial support
WORD COUNT: 1488
CORRESPONDING AUTHOR: Ziv Beckerman, MD Dell Children’s Medical Center, Center for Pediatric and Congenital Heart Disease, 4900 Mueller Blvd, Suite 3S.003, Austin, TX 78723 (zbeckerman@ austin.utexas.edu)
2
Abstract Bartonella endocarditis can be a very elusive diagnosis. The clinical manifestations can vary and, at times, include multiorgan involvement. This case report describes two patients presenting with multiorgan failure, cerebral mycotic aneurysms and valvular endocarditis secondary to Bartonella infection. The complex diagnosis, decision making, and surgical management are described.
3
This report describes two patients admitted to our institution one month apart of each other. Both had a very similar clinical presentation. Their respective case reports are described below, followed by a discussion.
Case Reports Case 1 The first patient is a 21-year-old male with a history of a bicuspid aortic valve. He had previously undergone balloon aortic valvuloplasty at the age of 9 years. He presented to our emergency department with high grade fever (39°C), acute renal failure, proteinuria, anemia and elevated Brain Natriuretic Peptide (BNP) (1453 pg/ml). His pertinent history included a 1-year history of night sweats, 2-months history of dry cough and 1-month history of bilateral ankle leg swelling. Transthoracic echocardiography revealed a thickened and dysplastic aortic valve, moderate to severe aortic insufficiency, mild aortic valve stenosis and moderately reduced left ventricular systolic function. No vegetations were seen. Concurrent work-up and treatment of both heart failure (with diuresis, inotrope infusion, and red blood cell infusion), renal failure, and suspected endocarditis were initiated. Broad spectrum empiric antibiotic coverage was started. His endocarditis work-up revealed Bartonella henselae on blood DNA testing. On a more focused anamnesis, the patient did report exposure to cats a few months previously. All other cultures came back negative. In addition, he underwent cerebral and cardiac Magnetic Resonance Imaging (MRI). His cerebral MRI demonstrated a small mycotic aneurysm, 2.5x2mm in the right middle cerebral artery (M3) distribution (Figure 1.). His cardiac MRI revealed a thickened bicuspid aortic valve with mild stenosis and severe regurgitation. Several multidisciplinary
4
discussions took place and the decision was made to address his mycotic brain aneurysm prior to surgical repair of his heart. Accordingly, he underwent cerebral angiography (Figure 2.) and following that, MRI-guided stereotactic right frontotemporal craniotomy and clip ligation of a mycotic aneurysm. Four weeks following his neurosurgery, he underwent aortic root replacement with a 24 mm aortic homograft valved conduit and closure of a patent foramen ovale via median sternotomy on cardiopulmonary bypass support. The operative findings were notable for a degenerated and necrotic aortic valve and aortic root abscess extending into the annulus of the right coronary artery cusp. Cross-clamp time was 174 minutes. Cardiopulmonary bypass time was 250 minutes. His postoperative recovery was uneventful, and he was discharged home 8 days after surgery. He completed 6 weeks of intravenous antibiotic treatment and continued doxycycline oral treatment for life. Case 2 The second patient is a 20-year-old male with a medical history of Noonan syndrome, von Willebrand disease, and asthma. He underwent pulmonary valve balloon valvuloplasty when he was 1 year of age. He presented with new onset symptoms of chest pain and shortness of breath. He was also found to have renal failure, severe proteinuria, BNP >5800 pg/ml, and anemia. His chest X-ray demonstrated pulmonary edema. Echocardiography demonstrated the mitral valve to be thickened with mild insufficiency and severe stenosis (mean pressure gradient of 1718mmHg), good biventricular function, and no echocardiographic evidence of vegetations. The infectious disease workup was positive for Bartonella vinsonii with blood DNA testing. Cerebral MRI demonstrated a 1.3 cm x 1 cm partially thrombosed left frontal middle cerebral artery mycotic aneurysm. Accordingly, he underwent a cerebral catheterization followed by a CTguided stereotactic left frontoparietal craniotomy for resection and ligation of the aneurysm. We
5
waited 4 weeks for him to recover from his neurosurgical intervention and proceeded with the cardiac repair. He underwent mitral valve replacement with a mechanical 29mm prosthesis. Operative findings were remarkable for congested lungs and engorged lymphatics. The left atrial wall was extremely thickened, the mitral valve itself was severely diseased with a miniscule orifice of 8-10 mm and thickening of the leaflets. There was obvious infection involving the anterior and posterior mitral valve leaflets. Cardiopulmonary bypass time was 171 minutes. Aortic cross-clamp time was 143 minutes. He was extubated on postoperative day-1 and did very well following that. He continued intravenous antibiotic treatment per the recommended protocol and continues oral doxycycline. His postoperative course was notable for a chylus pericardial effusion requiring drainage. Comment These two cases describe a unique presentation of Bartonella endocarditis with concurrent cardiac valvular disease and mycotic brain aneurysms. The initial presentation of both patients with heart failure, renal failure and stigmata of chronic illness while having an underlying cardiac pathology should alert the clinician for the possibility of an atypical endocarditis. The diagnosis of Bartonella endocarditis can be very elusive. Even though both patients had no evidence of vegetations by echocardiography, we elected to pursue all avenues to diagnose/rule out endocarditis. PCR-based diagnosis of Bartonella endocarditis and/or of other pathogens from blood has not been the standard recommended pathway for diagnosis of endocarditis neither in the adult or pediatric guidelines [1, 2]. That said, if we had not performed that assay, we would have been unlikely to identify the causative pathogen and would probably have not performed brain imaging. In that case, we would have potentially exposed those patients for neurological
6
complications secondary to their brain aneurysms during cardiac surgery. Additionally, neither of the patients would have received appropriate antibiotic coverage prior to their surgery. With regards to conduit/valve choices, undertaking an elaborate discussion with the patient and his family is paramount, as various surgical options, valves, and conduits exist. The first patient chose not to undergo a Ross procedure, and the second, chose a mechanical prosthesis after discussing all available surgical solutions (including a bioprosthesis with a potential for a future valve-in-valve option). The management of these patients is complex and requires a multidisciplinary approach to appropriately coordinate their care. Routine brain imaging is part of our workup for endocarditis, despite it being an area of controversy [3]. After diagnosing the brain aneurysms, we elected to address the cerebral issues first due to the concern of suffering a cerebral hemorrhage during open-heart surgery. Should an MRI of the brain be obtained in all infective endocarditis patients (or maybe only those with left-heart involvement) because cerebral emboli are so commonplace? No current consensus exists. Additionally, we had the concern of implanting a prosthetic cardiac valve while potentially having an infectious source elsewhere in the body. The decision to delay cardiac surgery in the setting of heart failure and acute valvular pathology can be questioned. Operating on the brain first results in a significant delay in the cardiac surgical care. The wait-time between the neuro- and cardiac surgeries, should be discussed by a multidisciplinary team, taking into consideration the pathogen’s behavior, the neurosurgical findings, the cardiac manifestations, and the clinical stability of the patient. These patients should remain in the intensive care unit as they can decompensate unexpectedly. If the patients are
7
deemed stable enough to undergo neurosurgical intervention first, it may be in their overall benefit for the long run. Additionally, addressing the cerebral issues first does allow for a longer course of antibiotic therapy prior to cardiac surgery. To conclude, (1) We advocate for extensive endocarditis evaluation including blood DNA testing whenever clinical suspicion exists even in the absence vegetations on echocardiography, (2) Diagnosis of endocarditis warrants cerebral imaging in our practice, (3) When clinically appropriate, the neurological findings should be addressed prior to performing a cardiac operation, and (4) A multidisciplinary approach is essential for caring of this complex patient group.
8
References 1. Baltimore RS, Gewitz M, Baddour LM, Beerman LB, Jackson MA, Lokhart PB, et al. Infective endocarditis in childhood: 2015 update. Circulation. 2015;132:1487–515. 2. Baddour L.M., Wilson W.R., Bayer A.S., Fowler V.G., Tleyieh I.M., Rybak M.J. Infective endocarditis in adults: diagnosis, antimicrobial therapy, and management of complications: a scientific statement for healthcare professionals from the American Heart Association. Circulation. 2015;132:1435–1486. 3. Champey J, Pavese P, Bouvaist H, Kastler A, Krainik A, Francois P. Value of brain MRI in infective endocarditis: a narrative literature review. Eur J Clin Microbiol Infect Dis. 2016;35:159–168.
9
Figure Legends Figure 1. Cerebral MRI demonstrating a cerebral aneurysm with hemorrhagic involvement. Figure 2. Cerebral angiography, a mycotic cerebral aneurysm in the right middle cerebral artery distribution.