Tricuspid and Aortic Valve and Ventricular Septal Defect Endocarditis: An Unusual Presentation of Acute Q Fever

Tricuspid and Aortic Valve and Ventricular Septal Defect Endocarditis: An Unusual Presentation of Acute Q Fever

Ann Thorac Surg 2009;88:2027–9 CASE REPORT PAGNI ET AL Q FEVER ENDOCARDITIS 2027 root was reconstructed by sewing on the porcine sinus to the resec...

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Ann Thorac Surg 2009;88:2027–9

CASE REPORT PAGNI ET AL Q FEVER ENDOCARDITIS

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root was reconstructed by sewing on the porcine sinus to the resected area of the sinus of Valsalva aneurysm (Fig 2D). The patient’s aortic valve was not distorted on visual inspection after reconstruction. The fistula tract area of the tricuspid valve was plicated with pericardium reinforced 4-0 Prolene sutures (Ethicon Inc, Somerville, NJ). The right atrial incision was closed primarily. Intraoperative transesophageal echocardiogram demonstrated preserved aortic root geometry and wellfunctioning aortic valve with trace aortic insufficiency (Fig 3). The tricuspid valve also demonstrated no regurgitation. The patient is doing well with normally functioning native aortic valve on a follow-up transthoracic echocardiographic study performed 50 days after surgery.

Comment

Valsalva aneurysm may cause sinus of Valsalva distortion, especially in patients with extensive aneurysms. A 38-year-old man presented to a local emergency room with dyspnea for 10 days. He had no significant past medical history, except for a surgically corrected congenital cleft palate. He was noted to have an aneurysm of the sinus of Valsalva and was transferred to our hospital. With further evaluation, he was found to have a ruptured sinus of Valsalva aneurysm. The aneurysm involved the noncoronary sinus, and it ruptured resulting in the formation of a fistulous communication to the right atrium (Fig 1). The aortic valve was normal, except for trivial regurgitation. The patient underwent surgical repair. A median sternotomy incision was made. Cardiopulmonary bypass was established by placing an arterial cannula in the ascending aorta and venous cannulae in the cavae. The ascending aorta was cross-clamped, and the cardioplegia solution was delivered in both the antegrade and retrograde fashion. The aorta was incised to expose the sinus of Valsalva and aortic valve. The valve had three equal-sized leaflets, and they coapted well. The aneurysm involved the noncoronary sinus with a fibrous fistula tract into the right atrium just above the tricuspid valve (Figs 2A and 2B). The aneurysmal sinus was resected. The noncoronary cusp of the aortic valve was left intact, suspended at the aortic annulus and commissures. Reconstruction of the sinus preserving the precise inter-commissural distance was challenging. We measured the size of the sinotubular juncture by utilizing the Freestyle porcine valve sizer (Medtronic Inc, Minneapolis, MN). One third of the circumference should have corresponded to the intercommissural distance of the intact sinus. The patient’s sinotubular juncture corresponded to a 25-mm Freestyle valve sizer (Medtronic Inc). We resected a segment of porcine aorta, including the noncoronary cusp attachment at the annulus and commissures. Then, the porcine aortic cusp was removed as well (Fig 2C). This resembled the needed sinus of Valsalva of the patient. The patient’s aortic © 2009 by The Society of Thoracic Surgeons Published by Elsevier Inc

The main method of surgical repair of ruptured sinus of Valsalva aneurysm has been resection of the aneurysm and patch repair of the aortic root using patch materials such as Dacron (DuPont, Wilmington, DE) or pericardium [1–4]. Reconstructing the sinus of Valsalva with a patch, without distorting the aortic valve competency, is important [1, 2], but it has been challenging because the aortic root geometry is very complex to reproduce. We have successfully utilized a porcine aortic sinus as a patch material to reconstruct the aortic root after sinus of Valsalva aneurysm resection. This seems to be a simple and reproducible method of reconstructing the geometry of sinus of Valsalva and aortic competency.

References 1. Van Son JAM, Danielson GK, Schaff HV, et al. Long term outcome of surgical repair of ruptured sinus of Valsalva aneurysm. Circulation 1994;90(Suppl 2):20 –9. 2. Jung SH, Yun TJ, Im YM, et al. Ruptured sinus of Valsalva aneurysm: transaortic repair may cause sinus of Valsalva distortion and aortic regurgitation. J Thorac Cardiovasc Surg 2008;135:1153– 8. 3. Feldman DN, Roman MJ. Aneurysms of the sinuses of Valsalva. Cardiology 2006;106:73– 81. 4. Azakie A, David TE, Peniston CM, Rao V, Williams WG. Ruptured sinus of Valsalva aneurysm: early recurrence and fate of the aortic valve. Ann Thorac Surg 2000;70:1466 –71.

Tricuspid and Aortic Valve and Ventricular Septal Defect Endocarditis: An Unusual Presentation of Acute Q Fever Sebastian Pagni, MD, Anthony Dempsey, MD, and Erle H. Austin III, MD Division of Thoracic and Cardiovascular Surgery, Department of Surgery, University of Louisville, Louisville, Kentucky

Q fever is a rare systemic infection caused by Coxiella Burnetii. The presentation with endocarditis is insidious, Accepted for publication April 15, 2009. Address correspondence to Dr Pagni, Division of Thoracic and Cardiovascular Surgery, University of Louisville, 201 Abraham Flexner Way, Suite 1200, Louisville, KY 40202; e-mail: [email protected].

0003-4975/09/$36.00 doi:10.1016/j.athoracsur.2009.04.141

FEATURE ARTICLES

Fig 3. Postoperative echocardiographic image demonstrating reconstructed sinus of Valsalva with good leaflet coaptation.

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CASE REPORT PAGNI ET AL Q FEVER ENDOCARDITIS

Ann Thorac Surg 2009;88:2027–9

with negative blood cultures, and oftentimes it is not obvious in diagnostic imaging studies until hemodynamic changes or valve destruction is reached [1]. We report a case of Q fever endocarditis involving the tricuspid and aortic valves and a congenital ventricular septal defect. Surgical treatment and distinct aspects of this unusual case are herein described. (Ann Thorac Surg 2009;88:2027–9) © 2009 by The Society of Thoracic Surgeons

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fever is a worldwide zoonosis caused by Coxiella Burnetii. Farmers and animal caretakers are more susceptible to this rare infection. Blood culture negative endocarditis is typically a common presentation with single valve involvement. Clinical diagnosis and surgical treatment are challenging.

FEATURE ARTICLES

A 39-year-old Caucasian man who is a farmer was referred to our institution with a 3-month history of malaise, fever, chills, and nonproductive cough. The symptoms worsened in the last month, with a new onset of myalgia and right pleuritic chest pain. His medical history revealed a ventricular septal defect (VSD) (Qp:Qs ⫽ 1.5:1), but he had not returned for follow-up since age 20. On presentation, his temperature was 37°C, his blood pressure was 120/75 mm Hg, and his heart rate was 80 bpm. His physical examination showed a 4/6 holosystolic murmur, best heard along the left sternal border. There was mild jugular vein distention and no signs of peripheral or cutaneous embolization. A micro-papular rash was noted in the lower extremities. The blood cultures at 5 days were negative and the white count was 6.9. A computed tomographic scan showed few small nodules in the right lung. A transesophageal echocardiogram (TEE) showed severe tricuspid regurgitation with papillary muscle rupture and multiple vegetations (Fig 1). A 1.2-cm vegetation was attached to the endocardium below the septal leaflet. A cardiac catheterism documented a perimembranous VSD with a Qp:Qs of 2.1:1 and pulmonary artery pressures of 43/22 mm Hg. His farming background and atypical

Fig 1. A 4-chamber transesophageal echocardiographic view shows the involvement of the tricuspid valve and subvalvar apparatus. A large vegetation sits on the atrial side of anterior tricuspid leaflet (arrow).

Fig 2. (A) Right atrial exposure with a view of the large ventricular septal defect (arrow), anterior leaflet vegetation, and endocardial vegetations after removal of the septal and part of the anterior leaflet of the tricuspid valve (*). (B) Tricuspid valve specimen shows multiple vegetations involving the leaflets, cordi, and septal papillary muscle.

presentation prompted serologic assays for rickettsiosis. Serology was positive for Coxiella Burnetti with titers of immunoglobular G phases I and II of 1/64 and positive for phase II immunoglobular M titers. At the operation (Fig 2), all tricuspid valve leaflets and subvalvular apparatus, VSD, and adjacent endocardium were involved with vegetations. Extensive debridement was performed, and the VSD was closed using autologous pericardium and a 31-mm bovine pericardial bioprosthesis (Carpentier-Edwards, Edwards Lifesciences LLC, Irvine, CA) was implanted. The results of an intraoperative transesophageal echocardiography also noted an 8-mm nodule on the undersurface of the noncoronary aortic leaflet. The mass was exposed through an aortotomy and had a healed fibrotic appearance; it was surgically shaved from the leaflet. An epicardial ventricular pacemaker lead was placed and was then postoperatively connected to an abdominal generator. The cardiopulmonary bypass time was 101 min and the cross clamp time was 86 min. Pathologic findings were inflammation changes with multiple histiocytoses of the leaflets and the vegetations, and the cultures were negative.

CASE REPORT CHINO ET AL INTIMAL TEAR AFTER ENDOVASCULAR REPAIR OF AORTIC DISSECTION

The patient’s recovery was uneventful; he was discharged home on postoperative day 9. He was treated for 12 months with oral doxycycline and hydrochloroquine. He was clinically free of reinfection at 3 years follow-up.

Comment Endocarditis is a very unusual but severe complication of Q fever that occurs in 75% of chronic Coxiella Burnetti infections and has an associated mortality of 25% [1]. Only approximately 2% of patients develop the chronic form of endocarditis that represents the principal complication. Coxiella endocarditis is rare, with only 12 cases reported in the United States in 2000 [2]. Patients with acute Q fever and pre-existing valvular or congenital defects and previous prosthetic valve replacement have an estimated 40% risk of developing endocarditis. The chronic presentation is nonspecific, manifesting with constitutional symptoms, valve dysfunction, and often heart failure. The presence of large vegetations or aggressive leaflet destruction, abscess, or fistulae are rare. Serology is the primary identification method for Coxiella Burnetti. Indirect immunofluorescence assay in acute Q fever may reveal a 4-fold increase in immunoglobular G titers between acute and convalescence samples or by the presence of phase II immunoglobular M antibodies [2]. Left-sided valves are more frequently involved, and the incidence is equally divided for both aortic and mitral valves. Mesana and colleagues [1] reported on 20 cases of Q fever endocarditis and only one case involved two valves. Tricuspid valve involvement has been reported in association with congenital coronary– cavitary fistula [3, 4], and VSD involvement was reported in only one report [5]. First-line antibiotic treatment consists of doxycicline (200 mg twice a day) and hydrochloroquine (200 mg three times a day) for a variable period of 1 to 3 years [6]. The risk of reinfection is approximately 8% for the aortic valve, as described by Agnihotri and colleagues [7]; however, it is unknown for the tricuspid valve or VSD locations. The presentation with multiple valve and VSD involvement in this case most likely represented aggressive infection in anatomically contiguous structures. The principles of surgical treatment of endocarditis were applied, including debridement and valve replacement. This case also involved debridement of the right ventricular endocardium and VSD rim, and resection of a tricuspid valve papillary muscle and aortic vegectomy. We conclude that Coxiella Burnetti infection should be highly suspected in cases of blood cultures negative for endocarditis. The association of extensive Q fever endocarditis (VSD, right ventricular endocardium, tricuspid and aortic valves) is extremely rare, and the diagnostic and surgical approach are challenging. Long-term antibiotic therapy is mandatory.

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2. Gami AS, Antonios VS, Thompson RL, Chaliki HP, Ammash NM. Q fever endocarditis in the United States. Mayo Clin Proc 2004;79:253–7. 3. Lupoglazoff JM, Brouqui P, Magnier S, Hvass U, Casasoprana A. Q fever tricuspid valve endocarditis. Arch Dis Child 1997;77:448 –9. 4. Hvass U, lansac E, Chatel D, Henri I. Mitral homograft for tricuspid valve endocarditis complicating a congenital fistula between the right coronary artery and right ventricle. J Heart Valve Dis 1996;5:564 – 6. 5. Laufer D, Lew PD, Oberhansli I, Cox JN, Longson M. Chronic Q fever endocarditis with massive splenomegaly in childhood. J Pediatri 1986;108:535–9. 6. Levy PY, Drancourt M, Etienne J, et al. Comparison of different antibiotic regimens for therapy of 32 cases of Q fever endocarditis. Antimicrob Agents Chemother 1991;35:533–7. 7. Agnihotri AK, McGiffin DC, Galbraith AJ, O’Brien MF. The prevalence of infective endocarditis after aortic valve replacement. J Thorac Cardiovasc Surg 1995;110:1708 –20.

Intimal Tear After Endovascular Repair of Chronic Type B Aortic Dissection Shuji Chino, MD, Noriyuki Kato, MD, Takatsugu Shimono, MD, and Kan Takeda, MD Departments of Radiology, and Thoracic and Cardiovascular Surgery, Mie University Hospital, Mie, Japan

Two patients with chronic type B aortic dissection underwent endovascular repair. The interval between the onset of aortic dissection and stent grafting was 1 year, 7 months in both patients. The entry closure was successful and postoperative course was uneventful for each patient. However, intimal injury developed at the bottom end of the stent graft 6 years after endovascular repair in 1 patient, and at 2 years in the other patient. The former patient underwent graft replacement of the descending thoracic aorta, and the latter underwent placement of additional stent grafts. (Ann Thorac Surg 2009;88:2029 –31) © 2009 by The Society of Thoracic Surgeons

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tent graft repair has become the first choice of the treatment in patients with a wide range of aortic diseases. Above all entry closure with endovascular repair, stent graft placement is now recognized as an attractive alternative to surgical intervention for the treatment of patients with aortic dissection. Stent grafting especially seems more promising in cases with chronic dissection, because aorta-related complications after the procedure rarely occur in these cases. In this report, we describe our experience of intimal injury after stent grafting in two cases with chronic aortic dissection.

References

Accepted for publication April 21, 2009.

1. Mesana TG, Collart F, Caus T, Salamand A. Q fever endocarditis: a surgical view and a word of caution. J Thorac Cardiovasc Surg 2003;125:217– 8.

Address corresponding to Dr Chino, Department of Radiology, Mie University Hospital, 2-174 Edobashi, Tsu, Mie, 514, Japan; e-mail: [email protected].

© 2009 by The Society of Thoracic Surgeons Published by Elsevier Inc

0003-4975/09/$36.00 doi:10.1016/j.athoracsur.2009.04.086

FEATURE ARTICLES

Ann Thorac Surg 2009;88:2029 –31