© 2013 Wiley Periodicals, Inc.
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Giant Candida Mycetoma in an Ascending Aorta Tubular Graft Giuseppe Di Benedetto, M.D., Rodolfo Citro, M.D., Antonio Longobardi, M.D., Generoso Mastrogiovanni, M.D., Antonio Panza, M.D., Severino Iesu, M.D., and Eduardo Bossone, M.D. Heart Department, University Hospital ‘‘San Giovanni di Dio e Ruggi d’Aragona’’, Salerno, Italy ABSTRACT We report the case of a 46-year-old male hospitalized for abdominal pain and fever with history of a David procedure followed by an aortic valve replacement due to severe aortic regurgitation. Transesophageal echocardiography (TEE) and computed tomography showed a large mass floating in the aorta. After surgical excision of the vegetation, attached to the Dacron prosthesis, histological examination revealed Candida hyphae and spores confirming the diagnosis of a mycetoma in an ascending aorta tubular graft. At six-month follow-up, the patient was in good clinical condition without recurrence of the fungal mass on TEE. doi:
10.1111/jocs.12193 (J Card Surg 2013;28:557–560) Fungal infections causing aortitis are increasingly diagnosed because of the growing number of immunocompromised patients undergoing cardiac surgery. Early- or late-onset infection complicates prosthetic aortic graft insertion in 0.5% to 5% of cases and is associated with considerable morbidity and mortality.1 We report a rare case of giant mycetoma attached to an ascending aorta tubular graft. PATIENT PROFILE A 46-year-old male was admitted with abdominal pain and fever. When he was 38 years old, he underwent a David procedure with a 28-mm Gelweave Valsalva Dacron conduit (Terumo Cardiovascular Systems Corp., Ann Arbor, MI, USA) because of an aortic root aneurysm associated with a bicuspid aortic valve. Two months before this admission, he underwent aortic valve replacement with implantation of a mechanical prosthesis (19 mm; St. Jude Medical, St. Paul, MN, USA) because of severe aortic regurgitation with dyspnea on exertion. The Dacron tube was left in place. Physical examination was unremarkable. Body temperature was 37.58C, no neurological disorders were observed, and blood tests were within the normal range. Abdominal computed tomography (CT) revealed splenic infarcts and Conflict of interest: The authors acknowledge no conflict of interest in the submission. Address for correspondence: Rodolfo Citro, M.D., F.E.S.C., University Hospital ‘‘San Giovanni di Dio e Ruggi d’Aragona’’, Heart Tower Room 810, Largo Citta` di Ippocrate, Salerno 84131, Italy. Fax: þ39 089 673314; e-mail: [email protected]
multiple abscesses along with multiple small cutaneous lesions on the upper extremities, compatible with systemic emboli. Blood cultures were positive for Candida albicans. Transesophageal echocardiography (TEE) showed normal function of the mechanical aortic prosthesis and a large mobile mass in the thoracic aorta (Fig. 1A–B). Urgent CT angio confirmed the presence of the mass (Fig. 1C–D). Multiple splenic infarcts were also documented. Amphotericin B was administered, and a staged surgical approach was planned. First, the patient underwent open splenectomy, followed the next day by surgical resection of the ascending aorta and excision of the mass (Fig. 2) with replacement of both the vascular prosthesis and aortic hemiarch. The vascular prosthesis was transected 5 mm above the coronary ostia and removed. A new 28-mm Dacron vascular prosthesis was implanted. The mycetoma formed within the ascending aortic prosthesis at the level of the sinotubular junction at the time of aortic valve replacement. The mechanical valve was not involved and did not require replacement. The mycetoma was large in size (14 cm 1.8 cm; Fig. 3). The histology revealed a large granuloma composed of Candida hyphae and spores (Fig. 4). Postoperatively, the patient developed a right pleural effusion, which was managed by thoracentesis. Cultural examination was positive for Candida spp. Subsequently, he developed an anaphylactic reaction to amphotericin B, and treatment with caspofungin 50 mg/day was started. In the following weeks, fluconazole 6 mg/kg/day was administered according to antimicrobial sensitivity studies. Lifelong oral antifungal therapy with fluconazole was recommended. At six-month follow-up, the patient was in good clinical condition without recurrence of the
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DI BENEDETTO, ET AL. CANDIDA MYCETOMA OF THE ASCENDING AORTA
J CARD SURG 2013;28:557–560
Figure 1. Transesophageal echocardiographic cross-sectional view at 08 (A) and longitudinal view at 938 (B) of the descending aorta showing the large free-floating fungal mass within the vessel lumen without evidence of the attaching site. C: Oblique sagittal CT image showing the vegetation arising about 3 cm above the aortic valve plane, extending to the descending thoracic aorta. D: Axial contrast-enhanced CT image clearly demonstrating mycetoma into the aortic arch.
Figure 2. Intraoperative view of the fungal vegetation attached to the Dacron prosthesis.
J CARD SURG 2013;28:557–560
DI BENEDETTO, ET AL. CANDIDA MYCETOMA OF THE ASCENDING AORTA
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Figure 3. Mycetoma dimensions (14 cm 1.8 cm) following surgical excision.
fungal mass at TEE. No prosthetic valve dysfunction was observed. IRB approval was obtained for this case presentation. DISCUSSION Damage to the aortic wall during the previous surgical procedure seems to be the underlying cause for the development of the fungal mass. It is believed that
fungal spores need wall damage in order to germinate and cause infection.1–3 Furthermore an aortic aneurysm developing from a weakness in the aortic wall can also lead to fungal infection along with diabetes mellitus, immune system impairment, and long-term antibiotic therapy.2,4 Mycotic aneurysm is associated with a worse prognosis because of the risk of vessel rupture, usually resulting in fatal bleeding. Similar cases of fungal
Figure 4. Hematoxylin and eosin stained histological sections showing Candida hyphae and spores (100).
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vegetation on a Dacron prosthesis due to Aspergillus flavus infection have been reported.5 The diagnosis is usually challenging. In patients with a recent history of aortic surgery, fever associated with embolic phenomena, with or without positive blood cultures, should raise the suspicion of mycetoma. In order to avoid dangerous diagnostic delays or misdiagnosis, it is worth noting that negative blood cultures do not exclude the possibility of fungal infection.4 Echocardiography is the first-line imaging modality to detect fungal vegetations. Additionally, echocardiography can predict embolic risk according to size and mobility of the mass.6 In our patient, the size and mobility of the vegetation supported the need for early intervention rather than a conservative approach. Fungal vegetations tend to be larger than those caused by bacterial infections and are associated with an increased embolic risk.2,7 In our case, CT provided evidence of multiple splenic infarcts. The appropriate approach to splenic infarction remains to be clearly elucidated.8 In view of the presence of fungal emboli, we decided to perform splenectomy prior to or at the time of valve replacement to prevent reinfection of the valve prosthesis.9 Combined treatment with aggressive antifungal therapy followed by surgery is the preferred method of treatment in patients with fungal prosthetic infections.10 Lifelong prophylactic therapy remains controversial, in particular for patients with fungal infection undergoing surgical treatment. In our case, long-term therapy combined with close follow-up was the most appropriate strategy.11
J CARD SURG 2013;28:557–560
REFERENCES 1. Motloch LJ, Rottlaender D, Darabi T, et al: Conservative management of Candida infection of prosthetic aortic graft by means of caspofungin and fluconazole alone. Tex Heart Inst J 2011;38:197–200. 2. Huang J, Bouvette MJ, Hagi Y, et al: Flow impeding fungal thrombus in the ascending aorta. Ann Thorac Surg 2008;86:1373–1375. 3. Middleton J, Chmel H, Tecson F, et al: Aortotomy site infections: Case presentation and review of the literature. Am J Med Sci 1980;279:105–109. I, Merino JL, et al: Aspergillus 4. Sanchez-Recalde A, Mate aortitis after cardiac surgery. J Am Coll Cardiol 2003;41:152–156. 5. Wandschneider W, Deutsch M: Fatal fungal infection of an ascending aortic graft. Thorac Cardiovasc Surg 1995; 43:217–219. 6. Correale M, Ieva R, Rinaldi M, et al: Voluminous mycetoma in a newborn with Down syndrome: Role of echocardiography. Eur J Echocardiogr 2006;7:398–400. 7. Wijesekera NT, Sheppard MN, Mullen MJ: Candida endocarditis with mycotic pulmonary emboli following re-do Rastelli operation. Heart 2004;90:e34. 8. Naito R, Mitani H, Ishiwata S, et al: Infective endocarditis complicated with splenic abscess successfully treated with splenectomy followed by double valve replacement. J Cardiol Cases 2010;2:e20–e22. 9. Wang CC, Lee CH, Chan CY, et al: Splenic infarction and abscess complicating infective endocarditis. Am J Emerg Med 2009;27:1021.e3–1025. 10. Luciani GB, Casali G, Viscardi F, et al: Tricuspid valve repair in an infant with multiple obstructive Candida mycetomas. Ann Thorac Surg 2005;80:2378–2381. 11. Muehrcke DD, Lytle BW, Cosgrove DM: Surgical and long-term antifungal therapy for fungal prosthetic valve endocarditis. Ann Thorac Surg 1995;60:538–543.
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Giant Candida Mycetoma in an Ascending Aorta Tubular Graft Giuseppe Di Benedetto, M.D., Rodolfo Citro, M.D., Antonio Longobardi, M.D., Generoso Mastrogiovanni, M.D., Antonio Panza, M.D., Severino Iesu, M.D., and Eduardo Bossone, M.D. Heart Department, University Hospital ‘‘San Giovanni di Dio e Ruggi d’Aragona’’, Salerno, Italy ABSTRACT We report the case of a 46-year-old male hospitalized for abdominal pain and fever with history of a David procedure followed by an aortic valve replacement due to severe aortic regurgitation. Transesophageal echocardiography (TEE) and computed tomography showed a large mass floating in the aorta. After surgical excision of the vegetation, attached to the Dacron prosthesis, histological examination revealed Candida hyphae and spores confirming the diagnosis of a mycetoma in an ascending aorta tubular graft. At six-month follow-up, the patient was in good clinical condition without recurrence of the fungal mass on TEE. doi:
10.1111/jocs.12193 (J Card Surg 2013;28:557–560) Fungal infections causing aortitis are increasingly diagnosed because of the growing number of immunocompromised patients undergoing cardiac surgery. Early- or late-onset infection complicates prosthetic aortic graft insertion in 0.5% to 5% of cases and is associated with considerable morbidity and mortality.1 We report a rare case of giant mycetoma attached to an ascending aorta tubular graft. PATIENT PROFILE A 46-year-old male was admitted with abdominal pain and fever. When he was 38 years old, he underwent a David procedure with a 28-mm Gelweave Valsalva Dacron conduit (Terumo Cardiovascular Systems Corp., Ann Arbor, MI, USA) because of an aortic root aneurysm associated with a bicuspid aortic valve. Two months before this admission, he underwent aortic valve replacement with implantation of a mechanical prosthesis (19 mm; St. Jude Medical, St. Paul, MN, USA) because of severe aortic regurgitation with dyspnea on exertion. The Dacron tube was left in place. Physical examination was unremarkable. Body temperature was 37.58C, no neurological disorders were observed, and blood tests were within the normal range. Abdominal computed tomography (CT) revealed splenic infarcts and Conflict of interest: The authors acknowledge no conflict of interest in the submission. Address for correspondence: Rodolfo Citro, M.D., F.E.S.C., University Hospital ‘‘San Giovanni di Dio e Ruggi d’Aragona’’, Heart Tower Room 810, Largo Citta` di Ippocrate, Salerno 84131, Italy. Fax: þ39 089 673314; e-mail: [email protected]
multiple abscesses along with multiple small cutaneous lesions on the upper extremities, compatible with systemic emboli. Blood cultures were positive for Candida albicans. Transesophageal echocardiography (TEE) showed normal function of the mechanical aortic prosthesis and a large mobile mass in the thoracic aorta (Fig. 1A–B). Urgent CT angio confirmed the presence of the mass (Fig. 1C–D). Multiple splenic infarcts were also documented. Amphotericin B was administered, and a staged surgical approach was planned. First, the patient underwent open splenectomy, followed the next day by surgical resection of the ascending aorta and excision of the mass (Fig. 2) with replacement of both the vascular prosthesis and aortic hemiarch. The vascular prosthesis was transected 5 mm above the coronary ostia and removed. A new 28-mm Dacron vascular prosthesis was implanted. The mycetoma formed within the ascending aortic prosthesis at the level of the sinotubular junction at the time of aortic valve replacement. The mechanical valve was not involved and did not require replacement. The mycetoma was large in size (14 cm 1.8 cm; Fig. 3). The histology revealed a large granuloma composed of Candida hyphae and spores (Fig. 4). Postoperatively, the patient developed a right pleural effusion, which was managed by thoracentesis. Cultural examination was positive for Candida spp. Subsequently, he developed an anaphylactic reaction to amphotericin B, and treatment with caspofungin 50 mg/day was started. In the following weeks, fluconazole 6 mg/kg/day was administered according to antimicrobial sensitivity studies. Lifelong oral antifungal therapy with fluconazole was recommended. At six-month follow-up, the patient was in good clinical condition without recurrence of the
558
DI BENEDETTO, ET AL. CANDIDA MYCETOMA OF THE ASCENDING AORTA
J CARD SURG 2013;28:557–560
Figure 1. Transesophageal echocardiographic cross-sectional view at 08 (A) and longitudinal view at 938 (B) of the descending aorta showing the large free-floating fungal mass within the vessel lumen without evidence of the attaching site. C: Oblique sagittal CT image showing the vegetation arising about 3 cm above the aortic valve plane, extending to the descending thoracic aorta. D: Axial contrast-enhanced CT image clearly demonstrating mycetoma into the aortic arch.
Figure 2. Intraoperative view of the fungal vegetation attached to the Dacron prosthesis.
J CARD SURG 2013;28:557–560
DI BENEDETTO, ET AL. CANDIDA MYCETOMA OF THE ASCENDING AORTA
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Figure 3. Mycetoma dimensions (14 cm 1.8 cm) following surgical excision.
fungal mass at TEE. No prosthetic valve dysfunction was observed. IRB approval was obtained for this case presentation. DISCUSSION Damage to the aortic wall during the previous surgical procedure seems to be the underlying cause for the development of the fungal mass. It is believed that
fungal spores need wall damage in order to germinate and cause infection.1–3 Furthermore an aortic aneurysm developing from a weakness in the aortic wall can also lead to fungal infection along with diabetes mellitus, immune system impairment, and long-term antibiotic therapy.2,4 Mycotic aneurysm is associated with a worse prognosis because of the risk of vessel rupture, usually resulting in fatal bleeding. Similar cases of fungal
Figure 4. Hematoxylin and eosin stained histological sections showing Candida hyphae and spores (100).
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DI BENEDETTO, ET AL. CANDIDA MYCETOMA OF THE ASCENDING AORTA
vegetation on a Dacron prosthesis due to Aspergillus flavus infection have been reported.5 The diagnosis is usually challenging. In patients with a recent history of aortic surgery, fever associated with embolic phenomena, with or without positive blood cultures, should raise the suspicion of mycetoma. In order to avoid dangerous diagnostic delays or misdiagnosis, it is worth noting that negative blood cultures do not exclude the possibility of fungal infection.4 Echocardiography is the first-line imaging modality to detect fungal vegetations. Additionally, echocardiography can predict embolic risk according to size and mobility of the mass.6 In our patient, the size and mobility of the vegetation supported the need for early intervention rather than a conservative approach. Fungal vegetations tend to be larger than those caused by bacterial infections and are associated with an increased embolic risk.2,7 In our case, CT provided evidence of multiple splenic infarcts. The appropriate approach to splenic infarction remains to be clearly elucidated.8 In view of the presence of fungal emboli, we decided to perform splenectomy prior to or at the time of valve replacement to prevent reinfection of the valve prosthesis.9 Combined treatment with aggressive antifungal therapy followed by surgery is the preferred method of treatment in patients with fungal prosthetic infections.10 Lifelong prophylactic therapy remains controversial, in particular for patients with fungal infection undergoing surgical treatment. In our case, long-term therapy combined with close follow-up was the most appropriate strategy.11
J CARD SURG 2013;28:557–560
REFERENCES 1. Motloch LJ, Rottlaender D, Darabi T, et al: Conservative management of Candida infection of prosthetic aortic graft by means of caspofungin and fluconazole alone. Tex Heart Inst J 2011;38:197–200. 2. Huang J, Bouvette MJ, Hagi Y, et al: Flow impeding fungal thrombus in the ascending aorta. Ann Thorac Surg 2008;86:1373–1375. 3. Middleton J, Chmel H, Tecson F, et al: Aortotomy site infections: Case presentation and review of the literature. Am J Med Sci 1980;279:105–109. I, Merino JL, et al: Aspergillus 4. Sanchez-Recalde A, Mate aortitis after cardiac surgery. J Am Coll Cardiol 2003;41:152–156. 5. Wandschneider W, Deutsch M: Fatal fungal infection of an ascending aortic graft. Thorac Cardiovasc Surg 1995; 43:217–219. 6. Correale M, Ieva R, Rinaldi M, et al: Voluminous mycetoma in a newborn with Down syndrome: Role of echocardiography. Eur J Echocardiogr 2006;7:398–400. 7. Wijesekera NT, Sheppard MN, Mullen MJ: Candida endocarditis with mycotic pulmonary emboli following re-do Rastelli operation. Heart 2004;90:e34. 8. Naito R, Mitani H, Ishiwata S, et al: Infective endocarditis complicated with splenic abscess successfully treated with splenectomy followed by double valve replacement. J Cardiol Cases 2010;2:e20–e22. 9. Wang CC, Lee CH, Chan CY, et al: Splenic infarction and abscess complicating infective endocarditis. Am J Emerg Med 2009;27:1021.e3–1025. 10. Luciani GB, Casali G, Viscardi F, et al: Tricuspid valve repair in an infant with multiple obstructive Candida mycetomas. Ann Thorac Surg 2005;80:2378–2381. 11. Muehrcke DD, Lytle BW, Cosgrove DM: Surgical and long-term antifungal therapy for fungal prosthetic valve endocarditis. Ann Thorac Surg 1995;60:538–543.
