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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
Right
©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
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
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
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
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
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
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
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
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
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
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
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
169
DISCLOSURE 170
We all have no conflict of interest to declare.
171 172
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 1
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Figure 2
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Figure 3a
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Figure 3b
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Figure 4
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Figure 5