Case Report Aortic Dissection Associated with Campylobacter Aortitis Sarah J Bucknell, FRCS, Thuy Le, FRACP, John Amerena, FRACP, David G Hill, FRACS and Malcolm McDonald, FRACP, FRCPA Department of Cardiothoracic Surgery, Geelong Hospital, Geelong, Victoria, Australia Infective aortitis and associated mycotic aneurysms are uncommon clinical entities and most reported cases are associated with Salmonella species We report successful surgical and medical treatment of a mycotic aneurysm of the aorta which presented as acute pericarditis and may have been caused by Camplyobacter jejuni (Heart, Lung and Circulation 2000; 9: 88–91) Key words: aortic dissection, Camplyobacter jejuni, infective aortitis M ycotic aneurysms of the thoracic aorta are uncommon and usually carry a poor prognosis An earlier report has quoted a mortality rate exceeding 80% The key to survival appears to be prompt surgery and long-term antibiotic therapy.2 Most of the reported cases of mycotic aneurysm of the aortic arch are due to Salmonella infection.1,2 We report a case of mycotic aneurysm thought to be due to Campylobacter jejuni, which presented as acute pericarditis associated with dissection and severe aortic regurgitation Case Report A 41-year-old previously well army officer presented to the Emergency Department with a 48 h history of severe pleuropericardial chest pain Prior to this he was in excellent health, regularly running 5–10 km per day His background was unremarkable except for an illness characterised by diarrhoea, fever and repeated rigors during a visit to rural Indonesia a few months previously Initial examination revealed a fit 82 kg man who was normotensive and with a temperature of 37.3°C The main physical finding was a loud pericardial rub and electrocardiogram showed widespread ST elevation suggestive of pericarditis He had no clinical features of Correspondence: Sarah Bucknell, Department of Cardiothoracic Surgery, Geelong Hospital, Geelong, Victoria 3220, Australia Marfan’s Syndrome There was a mild neutrophilia and monocytosis on blood screen, serum electrolytes were within normal limits and erythrocyte sedimentation rate (ESR) was mm/h Chest radiograph showed a moderately increased cardiothoracic ratio, but normal cardiac contour and aortic arch A provisional diagnosis of viral pericarditis was made; he was treated with indomethacin and morphine with some resolution of the pain The following morning, routine transthoracic echocardiography revealed a dilated aortic root (4.4 cm) and ascending aorta (5.8 cm) with an intimal flap in the ascending aorta consistent with type A aortic dissection Severe aortic regurgitation was noted on Doppler studies but it was not possible to accurately visualise the aortic valve He remained haemodynamically stable and was transferred to the operating theatre for urgent surgery Median sternotomy was performed and a small amount of turbid pericardial fluid was noted The ascending aorta had dissected; it was dilated and very fragile The primary tear appeared to be just above the level of the aortic valve and there was an almost full circumferential dissection upwards involving the whole ascending aorta and extending into the arch The dissection had extended downwards to the level of the coronary arteries, but they appeared patent The aortic valve was bicuspid; both leaflets were nodular, thickened and partially fused at the commissures Cardiopulmonary bypass was Heart, Lung and Circulation 2000; commenced after cannulating the right atrium and right femoral artery The cross clamp was applied just proximal to the inominate artery, cardioplegia given and then the aortic valve excised A St Jude aortic valve mounted in a Dacron conduit (Getz Bros, Minneapolis, MN, USA) was used and the proximal anastomosis completed using buttressed ethibond mattress sutures (Johnson & Johnson, Summerville, NJ, USA) The proximal coronary arteries were dissected free with a surrounding button and reimplanted into the graft The two layers of the dissected distal ascending aorta were reinforced using interrupted prolene sutures (Johnson & Johnson) buttressed with teflon felt (Burd, Teupe, AZ, USA) The conduit was then sutured in situ and the suture line reinforced with fibrin glue Pathological study of the specimen confirmed aortic dissection through the media and mild thickening of the valve leaflets Unexpectedly the aorta showed changes suggestive of acute neutrophilic aortitis Large numbers of neutrophils were present in the intima and had infiltrated into the media and adventitia The intima was oedematous and there was proliferation of the myofibroblasts Although no microorganisms were identified on Gram-stain the appearances were suggestive of a mycotic dissecting aneurysm Serial blood cultures and microscopy and culture of faeces all proved to be negative but the patient had received intravenous cephazolin as routine surgical prophylaxis Serology was sent for enteropathic bacteria and he was started on ciprofloxacin 750 mg twice daily It was thought that his illness in Indonesia could have been bacterial enteritis with subsequent secondary aortitis Serology was performed for a variety of potential pathogens; the only positive was that for Campylobacter jejuni with levels of immunoglobulin (Ig)M 0.81 (positive), IgG 1.00 (positive) and IgA 0.39 (low positive) The patient had a trouble free postoperative period and went home after days He returned to most of his regular activities, except long distance running, in months After 18 months on oral Ciprofloxacin 750 mg twice daily, he remained free of symptoms and all signs of active infection Discussion Osler first used the term mycotic aneurysm in 1885 to define any localised dilatation of the arterial tree caused by infection.3 Infectious arteritis is well documented in the literature and may present with or without aneurysmal dilatation There is no report of mycotic dissection In 1923, Stengel and Walferth studied 217 patients with infective aortic aneurysms and found that the majority of infections occur in the abdominal aorta (56% S J Bucknell et al Aortic Dissection 89 abdominal vs 17% thoracic).4 There was an overall male predominance and about 80% of the infections of the aorta appeared to result from valvular infections (endocarditis) In the pre-antibiotic era, bacterial endocarditis and its complications were universally fatal The first survival of a patient with an infected aortic aneurysm was reported in 1962 by Sower and Whelan,5 but even in the early 1970s most of the patients died In recent years, there have been an increasing number of cases of infective aortitis with or without a coexistent aneurysm reported in the literature Salmonella aortitis has been commonly reported with most patients found to have pre-existing atherosclerotic disease or a congenital anomaly at the site of the subsequent aneurysm.6 Infection in a pre-existing aneurysm is uncommon, occurring in only 3% of aneurysms.7 Several risk factors for infective aortitis are now more common than in the past These include diagnostic or therapeutic arterial catheterisation, intravenous drug use, immunocompromised patients due to chronic or neoplastic disease and the rising number of patients with AIDS The bacteriology of these infections also appears to have changed Streptococcus pyogenes, Streptococcus pneumonia, and Staphylococci were the predominant organisms identified in the pre-antibiotic era, while now Staphylococcus aureus and the Salmonella species account for about 40% of the total.7 Reports of other less common organisms such as Listeria monocytogenes, Bacteroides fragilis, Clostridium septicum and Mycobacterium tuberculous have appeared in the literature, especially in immunocompromised hosts.8,9 Enteric Gram-negative rods seem to have greater virulence in this context with a significantly higher incidence of aneurysmal rupture than aneurysms due to Gram-positive cocci (72 vs 25%).10 Campylobacters belong to a distinct group of specialised Gram-negative bacteria, rRNA superfamily V1 The two most common species are Campylobacter jejuni and Campylobacter coli, which are the principal causes of Campylobacter enteritis Campylobacter fetus has a particular predilection for the vascular endothelium in immunocompromised patients and since 1971 nine documented cases of Campylobacter fetus sepsis associated with abdominal aortic aneurysm have been reported.11 In these patients the aneurysm was noted to increase in size rapidly and rupture was a common cause of death In the present patient it is possible Campylobacter jejuni was the pathogenic organism, although this was not proven on culture Most patients with aortitis present with a subacute illness, but the course is highly variable with the duration of symptoms ranging from day to months and an average of weeks before diagnosis In patients with 90 S J Bucknell et al Aortic Dissection bacterial thoracic aortitis, fever was noted in 80%, chest and or shoulder pain in 45%, back pain in 20% and abdominal pain in 20%.2 A high index of suspicion is required as illustrated in the present patient who presented with characteristic symptoms and signs of pericarditis Patients with infective aortitis often have a comorbid condition associated with immunocompromise such as diabetes mellitus, chronic renal failure, chronic alcoholism or rheumatoid arthritis Johansen and Devin found an associated condition in 24% of patients8 and similar figures have been quoted by other studies.2,7 The overall mortality from infective aortitis is 53% rising to 95% in patients treated medically.2 The mortality from a thoracic aneurysm is higher than from abdominal aneurysms (75 vs 47%) Early diagnosis improves survival but diagnosis of an infected aortic aneurysm can be difficult Leucocytosis has been reported in 57% of cases and blood cultures were positive in 83%.2 Traditionally, aortography was considered essential for diagnosis but has largely been superseded by echocardiography, computed tomography and magnetic resonance image (MRI), which have greater sensitivity and specificity Echocardiography (both transthoracic and transoesophageal) can confirm the diagnosis, show the localisation and extent of the dissection and demonstrate any aortic insufficiency or pericardial effusion.12 Advantages of echocardiography are that it is portable and quick and MRI may not be available in all centres Early surgical intervention has greatly increased survival The aim of surgical therapy is to remove the infected tissues and re-establish distal arterial flow This can be performed by either extra-anatomic bypass outside infected tissue planes or direct graft interposition In situ reconstruction is recommended for thoracic aneurysms Prosthetic grafts are usually used with or without valve replacement depending on the extent of the dissection and involvement of the valve Use of a homograft to replace the aortic valve is a possibility and would be advantageous in the presence of established infection It would be difficult in the emergency setting to plan the use of a homograft due to availability of a suitable graft from the tissue bank and also if the dissection extends into the arch a homograft would not be long enough Bactericidal antibiotics should be used preoperatively and residual infection adequately treated postoperatively Ampicillin with or without gentamicin,13 sulphamethoxazole, trimethoprim14 and third generation cephalosporins15 have all proven to be effective agents More recently, fluoroquinones such as ciprofloxacin16 have been seen to be effective but duration of therapy is uncertain Heart, Lung and Circulation 2000; Conclusion This case of aortic dissection appears to be secondary to infective aortitis due to Campylobacter infection This is based on the pathological finding of an extensive inflammatory infiltrate within the dissected wall of the aorta suggesting a mycotic dissecting aneurysm He had had a previous gastroenteric illness and had positive serology for Campylobacter This, however, could be purely coincidental Another possibility is that the dissection had occurred spontaneously and the inflammatory changes were due to a dissection of 48 h standing There was a mild neutrophilia but no high fever or other constitutional symptoms No bacteria were seen on histological examination, Gram stains and cultures all proved to be negative The pathological finding of neutrophilic aortitis has important management and prognostic implications Should this be treated as possible infective aortitis? If so, the patient requires long-term antibiotics, close monitoring of inflammatory parameters and would have an uncertain prognosis as Campylobacter can be difficult to eradicate If the aetiology is a spontaneous dissection in a patient with a bicuspid aortic valve no further treatment would be required References Wilson SE, Gordan HE, Van Wagner PB Salmonella arteritis Arch Surg 1978; 113: 1163–6 Oskoui R, Davis WA, Gomes MN Salmonella aortitis Arch Intern Med 1993; 153: 517–25 Osler W The Gulstonian lectures on malignant endocarditis BMJ 1885; 1: 204–5 Stengel A, Walferth CC Mycotic (bacterial) aneurysms of intravascular origin Arch Intern Med 1923; 31: 527–54 Sower ND, Whelan TJ Suppurative arteritis due to Salmonella Surgery 1962; 52: 851–9 Parsons R, Gregory J, Palmer DL Salmonella infections of the abdominal aorta Rev Infect Dis 1983; 5: 227–31 Gomes MN, Choyke PL, Wallace RB Infected aortic aneurysms: a changing entity Ann Surg 1992; 215: 435–42 Johansen K, Devin J Mycotic aortic aneurysms: a reappraisal Arch Surg 1983; 118: 583–8 Harvey MH, Strachnan CSL, Thom BT Listeria Monocytogenes: a rare cause of mycotic aortic aneurysm Br J Surg 1987; 71: 166–7 10 Bennett DF, Cherry JK Bacterial infection of aortic aneurysm A clinicopathological study Am J Surg 1967; 113: 321–6 S J Bucknell et al Aortic Dissection Heart, Lung and Circulation 2000; 11 Rutherford EJ, Eakins JW, Maxwell JG, Tackett AD Abdominal aortic aneurysm infected with Campylobacter fetus, subspecies fetus J Vasc Surg 1979; 20: 373–7 12 Erbel R, Daniel W, Visser C, Engberding R, Roelandt J, Rennollet H Echocardiography in diagnosis of aortic dissection Lancet 1989; Mar 4, 457–61 13 Oz MC, McNicholas KW, Serra AJS, Spagna PM, Lemole GM Review of Salmonella mycotic aneurysms of the thoracic aorta J Cardiovasc Surg 1989; 30: 99–103 91 14 Donabedian H Long term suppression of Salmonella aortitis with an oral antibiotic Arch Intern Med 1989; 149: 1452 15 Trairatvorakul P, Sriphojanart S, Sathapatayavongs B Abdominal aortic aneurysms infected with Salmonella: problems of treatment J Vasc Surg 1990; 12: 16–19 16 Cook AM, Christopoulos D Rupture of a nonaneurysmal Salmonella infected aorta Clin Radiol 1989; 40: 605–6 Invited Commentary The case described represents aortic dissection, which is more common in a patient with a bicuspid aortic valve It may or may not be a case of dissection complicating infective aortitis I am not sure that one can definitely say this is a mycotic aneurysm for the following reasons: Inflammatory changes would be possible in an aortic dissection of 48 hours standing; hence, neutrophils, oedema, etc noted by the pathologist After only one dose of intravenous antibiotic, if this case was secondary to infection, one may still have expected to see bacteria present on histological examination, even if gram stains and cultures were rendered negative by the antibiotic The absence of fever greater than 38°C, or other constitutional symptoms, would be unusual, if this case were related to a serious infection such as aortitis or mycotic aneurysm Positive serology for Campylobacter could be purely coincidental In conclusion, it is possible that the dissection described in this case report could be secondary to infection, but this is by no means definite Peter Skillington ... case described represents aortic dissection, which is more common in a patient with a bicuspid aortic valve It may or may not be a case of dissection complicating infective aortitis I am not sure... increasing number of cases of infective aortitis with or without a coexistent aneurysm reported in the literature Salmonella aortitis has been commonly reported with most patients found to have pre-existing... possible Campylobacter jejuni was the pathogenic organism, although this was not proven on culture Most patients with aortitis present with a subacute illness, but the course is highly variable with