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Fusobacterium necrophorum is known to cause a life-threatening septicemic condition known as Lemierre syndrome. This disease is characterized as septic thrombophlebitis of the internal jugular vein, with distant metastatic spread to lungs, joints, and bone.1 A lesser known strain common in the gut flora, Fusobacterium nucleatum, can cause portal vein thrombophlebitis leading to liver abscesses.1 This presentation is known as the gastrointestinal (GI) variant of Lemierre syndrome. We report a rare case of F. nucleatum septicemia with septic pylephlebitis and multiple liver abscesses mimicking liver metastases.
CASE REPORTA 62-year-old woman with a medical history of hypertension and heart failure presented with malaise, confusion, low-grade fever, and myalgia. She had left-sided cramping abdominal pain with decreased appetite and intermittent hematochezia in the previous 1–2 months.
On admission, the patient was hemodynamically stable. Physical examination was remarkable for tenderness to palpation in the right upper quadrant and epigastrium, without guarding or rebound. Laboratory studies showed a white blood cell count of 12 K/μL with 81% neutrophils, hemoglobin 8.8 g/dL, mean corpuscular volume 78, alkaline phosphatase 128 U/L, creatinine 1.3 mg/dL (baseline 0.6 mg/dL), and blood urea nitrogen 28 mg/dL.
Abdominal and pelvic computed tomography (CT) with intravenous contrast (Figure 1) demonstrated hepatic lesions up to 9 cm, subocclusive thrombus in the superior mesenteric and portal veins, enlarged para-aortic lymph nodes, and thickened colon consistent with chronic diverticulitis. An infectious workup was significant for blood cultures that grew F. nucleatum. Enoxaparin and ampicillin-sulbactam were initiated. Magnetic resonance imaging (MRI)/cholangiopancreatography (Figure 1) redemonstrated multiple predominantly cystic hepatic masses suspicious for abscess vs metastatic disease. Transthoracic echocardiogram was negative for any valvular vegetation. Malignancy workup was unrevealing and included chest CT, brain MRI, carcinoembryonic antigen, alpha-fetoprotein, and cancer antigen 19-9. Colonoscopy (Figure 2) showed a benign-appearing, intrinsic 10 cm long stenosis of the distal sigmoid that was traversed, 5 mm sessile polyp in the mid-sigmoid that was not resected, extensive diverticula, and internal hemorrhoids, with no observed masses.
Figure 1.: (A) Abdominal and pelvic CT with IV contrast showing 2 large hepatic abscesses (green arrows) up to 9 cm in size. (B) Abdominal MRI T2 showing the 2 large hepatic abscesses (blue arrows). (C) Abdominal and pelvic CT with IV contrast showing subocclusive thrombus in the portal vein (red arrow head). (D) Abdominal and pelvic CT with IV contrast showing thickening of the sigmoid colon wall (yellow arrow head) and colonic diverticula (encircled). CT, computed tomography; IV, intravenous; MRI, magnetic resonance imaging.
Figure 2.: Colonoscopy showing (A, B) extensive diverticula with a mixed opening in the entire colon and (C, D) moderate benign-appearing intrinsic 10 cm length stenosis in the distal sigmoid.
Interventional radiology performed aspiration and drain placement of the 2 largest hepatic lesions. Aspiration yielded purulent fluid consistent with abscess and negative for malignancy. Aspirate culture grew F. nucleatum. By day 12, her abdominal pain had improved significantly and she was switched to ertapenem and apixaban and then discharged.
She continued to follow-up with infectious disease with abdominal CT every 4 weeks. After 6 weeks, drains were removed and she was switched to amoxicillin-clavulanate for an additional 6 weeks. She continued anticoagulation for a total of 12 weeks. An abdominal and pelvic CT scan (Figure 3) after 12 weeks showed near-complete resolution of the hepatic abscesses and mild residual nonocclusive thrombus in the superior mesenteric and portal veins. She lost to follow up with the GI clinic.
Figure 3.: Repeat abdominal and pelvic computed tomography with intravenous contrast after 12 weeks showing (A) near-complete resolution of hepatic abscess on the right lobe (green arrow) and complete resolution of hepatic abscess on the left lobe and (B) near-complete resolution of thrombus in the portal vein (red arrow head).
DISCUSSIONPylephlebitis, suppurative thrombophlebitis of the portal vein, is a rare but life-threatening complication of intra-abdominal or pelvic infections.2 Fusobacterium species are among the infectious causes precipitating thrombosis, which can present as internal jugular thrombophlebitis in Lemierre syndrome and as portal and superior mesenteric vein thromboses in the GI variant of Lemierre syndrome. The thrombogenic ability of the Fusobacterium species is because of its ability to aggregate platelets and vascular compression from inflammation and edema.3
PubMed and Scopus databases were searched for case reports or case series in English reporting pylephlebitis complicated by liver abscess due to Fusobacterium up to January 2023. Keywords used in the search included Lemierre syndrome, GI-variant Lemierre syndrome, pylephlebitis, fusobacterium, and liver abscess. The search yielded 9 cases4–12 (Table 1). Including our case, the mean age was 51.2 years (19–69 years), with a 1:1 male-to-female ratio. Four cases4,5,9,10 were F. necrophorum; 3 cases6,11,12 along with our case were F. nucleatum; and 2 cases7,8 were of unspecified Fusobacterium species. In our literature review, all infections were monomicrobial and community-acquired. The source of infection was GI sources in 5 cases (4 diverticular diseases, 1 pancreatitis), an oropharyngeal source in 2 cases, and post-GI procedure sources in 2 cases. The source could not be identified in 1 case (Table 2). We believe that diverticulitis is the source in our case, based on the left lower abdominal pain before presentation, signs of chronic diverticulitis on CT, and narrowing in the sigmoid colon on colonoscopy, which was likely diverticular stricture. Recent literature has shown that patients with F. nucleatum bacteremia have an increased risk of colorectal cancer.13 We emphasize that age-appropriate cancer screening in GI-variant Lemierre syndrome, including colonoscopy, should be considered to rule out malignancy.
Table 1. - Demographic data from all 10 case reports included in the literature review Study Country Age (sex) Medical history Underlying hypercoagulable diseases Symptoms Laboratory findings Abdominal pain Fever LC AST (U/L) ALT (U/L) ALP (U/L) TB (mg/dL) Current study, 2022 United States 62 (F) HTN and HFrEF None Present Present Present Normal Normal High Normal Furuncuoglu et al, 20214 Turkey 54 (F) Pancreatic adenocarcinoma None Present Present Present High Normal High High Radovanovic et al, 20205 United States 69 (M) Asthma, BPH, and SCC of the tongue None Present Present Present Normal Normal High N/A Tariq et al, 20196 United States 48 (F) Roux-en-Y bypass None Present None Present High High High High Rahmati et al, 20177 United States 59 (F) Multiple sclerosis None Present Present Present High High High High Buelow et al, 20138 United States 65 (M) N/A None Present Present N/A N/A N/A N/A N/A Kröll and Sendi, 20129 Switzerland 34 (M) Healthy None Present Present Present N/A N/A N/A N/A Shahani and Khardori, 201110 United States 34 (M) Chronic pancreatitis None Present None Present Normal Normal Normal Normal Clarke et al, 200311 United Kingdom 19 (F) Healthy None Present Present Present N/A High High High Etienne et al, 200112 France 68 (M) TB None None Present No High High N/A N/AALP, alkaline phosphatase; ALT, alanine transaminase; AST, aspartate transaminase; BPH, benign prostatic hyperplasia; CT, computed tomography; F, female; HTN, hypertension; HFrEF, heart failure with reduced ejection fraction; LC, leukocytosis; M, male; N/A, not available; SCC, squamous cell carcinoma; TB, tuberculosis; TB, total bilirubin.
CT, computed tomography; MRI, magnetic resonance imaging; N/A, not available; PCR, polymerase chain reaction; PV, portal vein; PVT, portal vein thrombosis; SMV, superior mesenteric vein; SV, splenic vein; US; ultrasound; >, switch to.
Clinical diagnosis of pylephlebitis is often challenging, requiring confirmation of thrombus formation in the portal vein or its branches and positive blood cultures.14 Six cases in the review had positive blood cultures while 7 had positive liver aspirate cultures (Table 2). Patients typically presented with a triad of dull right upper quadrant or epigastric pain, fever, and leukocytosis. The most common enzyme abnormality in our review was alkaline phosphatase (60%) (Table 1).
A prompt multidisciplinary approach is required to treat pylephlebitis, including a combination of antibiotics, abscess drainage, and anticoagulants.14 Coverage for Fusobacterium with combinations of penicillin/beta-lactamase inhibitors, metronidazole, cephalosporins, carbapenems, or clindamycin should always be pursued.5 Most regimens included an initial 2–4-week course of intravenous therapy, followed by oral therapy.5 The duration of treatment in the reported cases was variable and ranged from 2-12 weeks (Table 2). Source control and abscess drainage with culture are recommended in cases of liver abscess to confirm etiology and guide therapy.2,6,15 Abscess drainage was performed in 8 of the 10 cases in our review.
The role and duration of anticoagulant use remain controversial.16 Some authors recommend the universal use of anticoagulation because of higher recanalization rates while others suggest the selective use of anticoagulation.16 Anticoagulants were used in 7 of 10 of the reviewed cases, and the duration varied from 2 to 6 months. There is no definitive rule for testing for hypercoagulable diseases (HCDs). All the cases did not report a history of HCD, and 1 case tested for underlying hypercoagulable disease. Follow-up imaging is usually performed to confirm the resolution of liver abscess and portal vein thrombosis. Follow-up imaging with ultrasound, CT, or MRI of the abdomen was pursued in 8 of 10 cases. Cavernous transformation of the portal vein, a complication of long-standing portal vein thrombosis, was noticed in 2 cases.
In conclusion, the GI variant of Lemierre syndrome can mimic metastatic liver lesions with portal vein thrombosis. Therefore, excluding malignancy and rapidly recognizing distinctive clinical features is critical. With a mortality rate of 11%–35%,2,14 early detection and prompt treatment of GI-variant Lemierre syndrome is crucial to delivering quality, patient-centered care.
DISCLOSURESAuthor contributions: F. Jaber: writing the draft and manuscript, project administration, and is the article guarantor. S. Alsakarneh: writing the manuscript and literature review. J. Campbell: reviewing and editing the manuscript and figure organization. A. Awad: writing the manuscript and organizing the table. WT Mohamed: reviewing and editing the manuscript and literature review. K. Wittler: reviewing and editing the manuscript and supervision. H. Ghoz: reviewing and editing the manuscript, resolving points of conflict, and supervision. W. Clarkston: reviewing and editing the manuscript and supervision.
Financial disclosure: None to report.
Previous presentation: This abstract was presented as a poster presentation at the American College of Gastroenterology (ACG) Meeting; October 24, 2022; Charlotte, North Carolina, and got an outstanding poster presenter award.
Informed consent was obtained from the patient.
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