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Endovascularly Treated Superficial Femoral ArteryAneurysm Rupture Secondary to Campylobacter fetusBacteremia: A Case Report

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Posted at the Institutional Resources for Unique Collection and Academic Archives at Tokyo Dental College, Available from http://ir.tdc.ac.jp/

Title

Endovascularly Treated Superficial Femoral Artery Aneurysm Rupture Secondary to Campylobacter fetus Bacteremia: A Case Report

Author(s)

Alternative Ono, S; Shimogawara, T; Hasegawa, H

Journal Annals of vascular surgery, 72(): 664.e1‑664.e6 URL http://hdl.handle.net/10130/5869

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©2021. This manuscript version is made available under the CC‑BY‑NC‑ND 4.0 license

https://creativecommons.org/licenses/by‑nc‑nd/4.0/

Description

(2)

Endovascularly treated superficial femoral artery aneurysm rupture secondary to 1

Campylobacter fetus bacteremia: A case report 2

3

Shigeshi Ono,1 Tatsuya Shimogawara,1 and Hirotoshi Hasegawa,1 4

5

1Department of Surgery, Tokyo Dental College Ichikawa General Hospital, Chiba, 6

Japan.

7 8

Correspondence to: Shigeshi Ono, MD, PhD, Department of Surgery, Tokyo Dental 9

College Ichikawa General Hospital, 5-11-13 Sugano, Ichikawa, Chiba, Japan; telephone:

10

81-47-322-0151; fax: 81-47-322-7931; E-mail:[email protected] 11

12

(3)

ABSTRACT:

13

Degenerative aneurysms of the superficial femoral artery (SFA) are relatively 14

rare and often recognized when they become symptomatic such as rupture. Infected SFA 15

aneurysms are much rarer, especially those caused by Campylobacter fetus 16

bacteremia. We report a case of a 67-year-old woman referred to our hospital owing to 17

the presence of a painful reddish swelling on her left thigh. A huge SFA aneurysm 18

rupture was diagnosed, and endovascular treatment with covered stent was performed.

19

C. fetus was detected in the blood culture thereafter, and antibacterial therapy was 20

successfully performed without any additional surgical interventions. She remained well 21

without any evidence of indolent infection 19 months after the endovascular treatment.

22

The endovascular approach with appropriate prolonged antibacterial therapy would be a 23

feasible alternative for managing selected infected aneurysm cases.

24 25

(4)

INTRODUCTION 26

Degenerative true aneurysms of the superficial femoral artery (SFA) are 27

relatively rare and usually not diagnosed until they reach a considerable diameter or 28

become symptomatic. Aneurysm rupture is one of the most common symptoms. We 29

present the case of a 67-year-old woman referred to our hospital with the diagnosis of left 30

thigh cellulitis, which turned out to be an infected SFA aneurysm rupture due to 31

Campylobacter fetus bacteremia.

32

Written informed consent was obtained from the patient for the publication of 33

this report and any accompanying images.

34 35

CASES REPORT 36

A 67-year-old woman was referred to our hospital due to the presence of 37

painful reddish swelling on her left thigh with the diagnosis of cellulitis. Eleven days 38

prior to the admission, she had low-grade fever and diarrhea. High fever up to 38.8 °C 39

had persisted intermittently thereafter and she had severe pain on her left thigh 3 days 40

(5)

prior to the admission. Her medical history included hypertension, hyperlipidemia, and 41

diabetes mellitus, and she is a current smoker.

42

On the day of admission, her temperature was 38.0°C, and the laboratory 43

findings were as follows: white blood cell count: 15,900/mm3, hemoglobin: 11.6 g/dL, 44

C-reactive protein: 20.57 mg/dL, procalcitonin: 0.28 ng/ml, and HbA1c: 7.3%. No 45

bacteria were detected in the blood culture. A physical examination confirmed the 46

presence of a subcutaneous hematoma throughout her left thigh (Fig. 1). A contrast- 47

enhanced computed tomography (CT) scan revealed a ruptured SFA aneurysm with a 48

mural thrombus of 58 mm in diameter and an extended hematoma (Fig. 2).

49

Contralateral SFA was within normal limits, and no abdominal aortic aneurysm was 50

observed.

51

We proceeded to aneurysm exclusion with a self-expandable covered stent 52

(GORE®, Viabahn®) (Fig. 3A, B). Intravenous antibiotics were administered with 53

sulbactam sodium/ampicillin sodium (SBT/ABPC) 3 g 4 times daily (QID) until 54

postoperative day 6. Her postoperative course was uneventful, inflammation markers 55

were improved, and she had been afebrile. She was discharged on postoperative day 8.

56

(6)

On postoperative day 17, she was readmitted to our hospital owing to high 57

fever (temperature up to 39.0°C), and a contrast-enhanced CT scan revealed the severe 58

inflammation around the covered stent (Fig. 4). Empiric treatment was initiated with 59

intravenous cefazolin (CEZ) 1 g 3 times daily. However, C. fetus was detected in the 60

blood culture, which is resistant to CEZ. Therefore, we immediately switched to 61

intravenous ABPC 2 g QID for 2 weeks. The level of inflammatory markers decreased 62

without any invasive management. A follow-up contrast-enhanced CT scan revealed 63

significantly improved inflammation (Fig.5), and antibiotics were switched to chronic 64

oral amoxicillin for 1 year. She remained well without any evidence of indolent 65

infection 19 months after the endovascular treatment.

66 67

DISCUSSION 68

The incidence of isolated degenerative femoral artery aneurysms is reported to 69

be approximately 0.005% (1). They represent only 3% of all peripheral aneurysms, and 70

80% of them have been reported to be in the common femoral artery, 15% in the SFA, 71

and 5% in the deep femoral artery (2-5). Most of SFA aneurysms are located in the 72

(7)

middle-distal third of the SFA. Therefore, SFA aneurysms are less likely to be palpable 73

and recognized, and the majority of them are not detected until they reach a considerable 74

diameter or patients exhibit any symptoms (3, 6). Moreover, an SFA aneurysm is usually 75

caused by iatrogenic or other traumas, or conditions related to inflammatory and 76

infectious processes, or connective tissue diseases (7, 8). Representative rheumatologic 77

diseases related to vasculitis or aneurysms include Bechet and nodular polyarteritis;

78

however, our case did not have stomatitis aphthosa, genital ulcers, and any skin diseases.

79

She also had no signs of other connective tissue disorders or vasculitis. In addition, the 80

rarity of an SFA aneurysm without any traumas or symptoms, such as rupture, acute 81

ischemia, or limb edema due to compression on the deep vein, makes the diagnosis much 82

challenging.

83

Since infected aneurysms were first described by Osler (9), they have been 84

reported to constitute up to 3% of aneurysms, and most of them are the abdominal aorta 85

and femoral artery aneurysms (10, 11). Immune status is considered to be an important 86

factor in the formation of an infected aneurysm (12). Atherosclerosis allows bacterial 87

colonization in the arterial intima, resulting in a local immune response and the activation 88

(8)

of T-cells, which release cytokines that increase protease activity (10, 13). The reduced 89

structural integrity of the artery, due to breakdown of elastin and collagen, has been 90

considered to result in the formation of an infected aneurysm (10, 13).

91

It is crucial to obtain a microbiological diagnosis for managing a critical course 92

of an infected aneurysm. Staphylococcus aureus is a common causative pathogen, and 93

enteric organisms such as Salmonella species, Escherichia coli, and Pseudomonas 94

aeruginosa have also been isolated (14-19).

95

As of now, more than 15 different Campylobacter species have been identified 96

(20). C. fetus, comprising two species, has been recognized as a major species causing 97

extraintestinal lesions (20) and bacteremia, particularly in elderly and 98

immunocompromised patients (21, 22). The most common clinical symptoms are 99

diarrhea and cellulitis; however, endovascular infection is serious and relatively frequent 100

compared to infection caused by other Campylobacter species (21, 22). C. fetus has been 101

reported to have a tropism for vascular endothelium or arterial destructive potential 102

particularly where preexisting damage is preexisting (23). Farm animals are considered 103

to be the major reservoir of Campylobacter species, and the major cause of bacteria- 104

(9)

related food poisoning and foodborne gastrointestinal infections. Our patient had no 105

contact with any animals and had not eaten any suspicious food, except for a properly 106

heated chicken dish before the onset of diarrhea. She also did not have any signs of 107

peripheral artery disease or aneurysms, and was not even under an immunocompromised 108

status, except for diabetes. The reason behind the bacterial colonization occurred in the 109

SFA still remains unclear.

110

Currently, complete resection of the infected aneurysm with adjacent tissues 111

and autologous reconstruction is the gold standard to manage infected aneurysms. The 112

prosthetic graft was considered to be avoided or resected if used. However, even if 113

aggressive debridement was performed, reinfection could occur at a relatively high rate 114

(24). Recently, the strategy of in situ preservation of the infected graft has also been 115

reported. (25-27). Endovascular aneurysm repair (EVAR) has been introduced as an 116

alternative for infected abdominal aortic aneurysms (IAAAs); this repair is particularly 117

essential in the case of severely ill or elderly patients. Endovascular treatment was 118

considered to be a temporary treatment followed by definitive open repair, which was 119

therefore called a “bridge to open surgery.” However a European multicenter trial showed 120

(10)

the durability of EVAR by assessing late infection-related complications and long-term 121

survival. The study concluded that EVAR can be a durable treatment option for most 122

patients (15). Additionally, another recent study showed acceptable long-term outcomes 123

of EVAR for IAAAs (16). Appropriate antibacterial therapy with suitable duration would 124

be the most definitive factor for this treatment.

125

C. fetus is typically very sensitive to ampicillin, third-generation 126

cephalosporins, aminoglycosides, imipenem, and meropenem (21, 28, 29); therefore, our 127

antibiotics alone strategy was acceptable even after the second admission with the 128

presence of a prosthetic device. As for the duration of postoperative antibacterial therapy 129

for infected aneurysms to prevent indolent infection, although some authors 130

recommended a minimum of 1–2 months, recent reports have claimed that 6 months or 131

even a lifelong use may be required (15, 30, 31). We consider that prolonged antibacterial 132

therapy is necessary, and oral amoxicillin was administered for 1 year. If S. aureus or 133

Candida species was the causative pathogen, the lifelong treatment with antibiotics may 134

be required. The duration should be considered depending on the pathogens, and 135

continuous follow-up should be done even after discontinuing the antibacterial therapy.

136

(11)

Arterial destruction is often observed in the presence of arterial infection, 137

especially in the presence of the prosthetic devices (32, 33), and even without the presence 138

of devices (34, 35). Biofilm constructed around the prosthetic devices may inactivate 139

antibiotics or would prevent antibiotics from penetrating, making infection control 140

difficult (36). However, depending on the bacteria or the materials of the prosthetic device, 141

antibiotics alone strategy could be a feasible option in selected cases (36-39).

142

We should have considered the potential indolent infection at the time of initial 143

treatment from several clinical signs, and should have used a prolonged antibiotic therapy.

144

The diarrhea 2 weeks before the first admission should have made us consider C. fetus 145

infection, which was resistant to CEZ. No bacteria were detected in the first blood culture;

146

therefore, deciding the appropriate antibiotics and their dosage and duration was difficult.

147

However, even though an infection was apparent in the first place, endovascular treatment 148

with appropriate antibacterial therapy could be a feasible alternative for managing 149

infected aneurysms.

150

We consider that an emergent case, such as ours, could be treated 151

endovascularly, and even in an elective case, it could be tried unless the bacteria is multi- 152

(12)

drug resistant. However, clinicians should be prepared to perform appropriate drainage 153

or surgical intervention in case the infection becomes uncontrollable. If a multi-drug 154

resistant bacterial infection was apparent in the first place, surgical repair should be the 155

first choice.

156

Another problem is that the use of a covered stent use for SFA or popliteal artery 157

aneurysms is not covered by the health insurance in Japan. Analysis of further cases will 158

be needed to ensure that kind of treatment is effective and feasible.

159

The findings from our case highlight the rarity of a C. fetus-infected aneurysm 160

and the efficacy of endovascular treatment with appropriate antibacterial therapy for 161

infected aneurysms.

162 163

CONCLUSION 164

We observed a rare case of C. fetus-infected SFA aneurysm rupture.

165

Endovascular treatment with prolonged antibacterial treatment might be a feasible 166

alternative to treat selected infected aneurysms.

167 168

(13)

169

DISCLOSURE 170

We all have no conflict of interest to declare.

171 172

(14)

FIGURE CAPTIONS 173

Fig. 1 A huge hematoma on the patient’s left thigh.

174 175

Fig. 2 Contrast-enhanced computed tomography scan shows a ruptured superficial 176

femoral artery (SFA) aneurysm with a mural thrombus of 58 mm in diameter and an 177

extended hematoma on the left thigh. Contralateral SFA was within normal limits.

178 179

Fig.3A The initial angiogram shows a ruptured superficial femoral artery aneurysm.

180 181

Fig. 3B The final angiogram shows a complete exclusion of the aneurysm.

182 183

Fig. 4 Contrast-enhanced computed tomography scan shows severe edema and 184

inflammation around the covered stent.

185 186

Fig. 5 Contrast-enhanced computed tomography scan shows significant 187

improvement in inflammation.

188 189

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Figure 5

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