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Sunday Poster Session

Category: Liver

P0907 - Gastrointestinal Variant of Lemierre's Syndrome: A Comprehensive Review and Analysis of 36 Case Reports

Sunday, October 22, 2023

3:30 PM - 7:00 PM PT

Location: Exhibit Hall

Presenting Author(s)

Fouad Jaber, MD

University of Missouri-Kansas City
Kansas City, MO

Fouad Jaber, MD¹, Saqr Alsakarneh, MD¹, Tala Alsharaeh, MS², Azizullah Beran, MD³, John Campbell, MD¹, Mohammad Jaber, MD⁴, Ahmed-Jordan Salahat, MS², Natalie Wilson, MD⁵, Yazan Abboud, MD⁶, Mohammad Almeqdadi, MD⁷, Laith Numan, MD, MS⁸, Nikki duong, MD⁹, Hassan Ghoz, MD¹, John Helzberg, MD¹, Wendell Clarkston, MD¹⁰
¹University of Missouri-Kansas City, Kansas City, MO; ²University of Jordan, Amman, 'Amman, Jordan; ³Indiana University, Indianapolis, IN; ⁴Al-Azhar University, Gaza, Palestinian Territories; ⁵University of Minnesota, Minneapolis, MN; ⁶Rutgers New Jersey Medical School, Newark, NJ; ⁷Lahey Medical Center, Boston, MA; ⁸Saint Louis University, St. Louis, MO; ⁹Virginia Commonwealth University, Richmond, VA; ¹⁰University of Missouri-Kansas City School of Medicine, Saint Luke’s Hospital of Kansas City, Kansas City, MO

Introduction: Lemierre's syndrome (LS) is typically caused by Fusobacterium necrophorum (F. necrophorum), and is characterized by internal jugular vein thrombophlebitis and septic emboli, predominantly affecting the lungs. A gastrointestinal (GI) variant of LS involving Fusobacterium species causing pylephlebitis and potential liver abscesses has been identified. This systematic review aims to summarize the currentliterature on the GI variant of LS.

Methods: A systematic search of PubMed, Scopus, and Cochrane databases was conducted up to February 2023 for case reports detailing the GI variant of LS. Data regarding demographics, clinical features, treatment, and outcomes were extracted. Descriptive statistics were employed, with means and standard deviations for continuous variables and percentages for dichotomous and categorical variables.

Results: 36 patients were included , with a mean age of 50.75 years, and male predominance of 72%. Cultures were positive for F. nucleatum, F. necrophorum, and negative in 47.2%, 33.3%, and 11.1% of cases, respectively. The primary source of infection was the GI tract (55.5%),mainly diverticular disease (25%). Patients typically presented with abdominal pain (72.2%), fever (86.1%), and leukocytosis (86.1%). Elevated alkaline phosphatase levels were observed in 61.1% of cases. Thrombosis primarily involved the portal veins (77.7%), followed by the superior mesenteric vein (30.5%). Liver abscesses were detected in 18 cases (50%), with positive cultures in 12 (92.3%). Treatment included penicillin/penicillin-lactamase inhibitors alone (17%), and ertapenem alone (17%). Abscess drainage was performed in 13/18patients, resulting in complete or near-complete abscess resolution in 83% of cases. Long-term anticoagulation therapy was administered in 27 cases (75%), predominantly using warfarin or vitamin K antagonists (59.2%). The outcomes of thrombosis were: resolution (19.4%), persistence (19.4%), and cavernous transformation (16.6%). No fatalities were reported in this cohort.

Discussion: GI variant of LS should be considered in patients presenting with abdominal pain, fever, leukocytosis, and thrombosis involving the portal or superior mesenteric veins. The identification of Fusobacterium species in blood cultures and the presence of liver abscesses further support the diagnosis. A multidisciplinary approach including antibiotic therapy, abscess drainage, and long-term anticoagulation is required in managing these patients.

Study

Demographics

Presenting Symptoms

Laboratory Findings on Presentation

Work-up

Treatment

Outcome

Age (Sex)

Abdominal Pain

Fever

LC
(Y/N)

AST/ALT/ALP/TB

Infection Source

Thromboses Site

Blood culture

Liver Abscess

Aspiration Culture

Abscess Drainage

Antibiotics

Anti- coagulation

Follow-up Imaging

(Duration)

Vein Thrombosis

Liver Abscess

Jaber, 2022

62 (F)

Yes

N/H/N/N

Diverticulitis

PV, SMV

F. nucleatum

Yes

F. nucleatum

Yes

I: Amp/sul
F: Ertapenem,

followed by

Amox/clav

( 12 weeks)

I: Enoxaparin
F: Apixaban
(12 weeks)

CT
(12 weeks)

Near-complete resolution

Mohammadian, 2022

86 (F)

Yes

H/H/H/H

Diverticular disease with abscess

SV, SMV, PV, Peroneal VT

F. nucleatum

N/A

I: Cefepime, metro
F: Amox/clav

(4 weeks)

I: Heparin
F: Apixaban
(N/A)

CT
(2 weeks)

Persistent thrombosis

N/A

Barker, 2022

72 (M)

Yes

H/H/N/N

Diverticulitis

PV, Popliteal vein

F. nucleatum,

F. periodonticum

Yes

F. periodonticum

Yes

I: Vanc, pipera/tazo
F: Amox/clav

(4 weeks)

I: Heparin
F: Apixaban
(N/A)

CT
(1 month)

N/A

Complete Resolution

Abbas, 2022

73 (M)

Yes

N/N/H/N

Unknown

SMV, PV

F. nucleatum

Yes

N/A

I: Pipera/tazo, metro
F: Ertapenem
(6 weeks)

I: Enoxaparin
F: Warfarin
(6 weeks)

None

N/A

Hitchcock, 2021

27(M)

Yes

H/H/H/N

Unknown

Negative

Yes

F. nucleatum

Yes

I: Ceftriaxone,

Azithromycin

followed by

pipera/tazo
F: Amox/clav

(6 weeks)

Warfarin
(6 weeks)

None

N/A

Lazar, 2021

57 (F)

Yes

H/H/N/N/A

Sigmoid Colitis

SMV, PV

F. necrophorum

N/A

I: Vanc, metro
F: Ertapenem
(2 weeks)

I: Heparin
F: Apixaban
(3 months)

None

N/A

Furuncuoglu, 2021

54 (F)

Yes

H/N/H/H

Surgical site

(Post-total pancreatectomy due to pancreatic adenocarcinoma)

F. necrophorum

Yes

N/A

Cefoperazone, sulbactam
(1.5 weeks)

None
(N/A)

MRI

Complete resolution

Cheng, 2021

71 (M)

Yes

H/H/H/N/A

Pancolitis

PV, SMV, IMV

F. nucleatum

N/A

I: Ceftriaxone, metro

F: Cefpodoxime,

metro followed by

Amox/clav
(3 weeks)

I: Enoxaparin
F: Rivaroxaban
(3 months)

CT venogram

(3 months)

Complete resolution

N/A

Tharu, 2020

41 (M)

Yes

N/N/H/N

Diverticular disease

Fusobacterium

Yes

F. nucleatum

Yes

I: Vanc, ceftriaxone,

metro
F: Ceftriaxone, metro
(6 weeks)

None
(N/A)

CT
(4 weeks)

Persistent thrombosis

Partial resolution

Abdallah, 2020

37 (M)

Yes

H/H/H/H

Diverticulitis

IMV

F. nucleatum,

B. fragilis,

C. fragilis

I: Meropenem

F: Ertapenem
(1 month)

I: Heparin
F: Warfarin
(6 months)

CT
(1 month)

Persistent thrombosis

N/A

Hammami, 2020

38 (F)

Yes

N/A

Presumed lower GI

F. necrophorum

Yes

N/A

I: Ceftriaxone, metro

F: Rifampicin, metro
(3 months)

None
(N/A)

CT
(3 months)

Complete resolution

Handa, 2020

39 (M)

Yes

H/H/H/H

Unknown

F. nucleatum

N/A

I: Ceftriaxone, metro

F: Metro
(2 weeks)

I: Enoxaparin
F: Apixaban
(3 months)

None

N/A

Radovanovic, 2020

69 (M)

Yes

N/N/H/N

Presumed oropharyngeal

F. necrophorum

Yes

F. necrophorum

Yes

I: Ceftriaxone, metro

F: Amox/clav
(5 weeks)

I: Enoxaparin
F: Warfarin
(N/A)

N/A

Tariq, 2019

48 (F)

Yes

H/H/H/H

Surgical site

F. nucleatum

Yes

F. nucleatum

Yes

Ceftriaxone, metro
(6 weeks)

I: Enoxaparin
F: Rivaroxaban
(6 months)

(4 months)

Persistent thrombosis and formation of collaterals

Complete resolution

Mellor, 2017

64 (M)

Yes

N/N/H/H

Diverticulitis and peri colonic abscess

IMV

Fusobacterium

N/A

I: Pipera/tazo
F: Ertapenem
(4 weeks)

Heparin
(Few days then discontinued)

(4 weeks)

Complete resolution

N/A

Moore, 2017

60 (M)

Yes

N/N/N/N

Presumed subclinical GI infection

F. nucleatum

N/A

I: Pipera/tazo
F: Metro
(4 weeks)

I: Enoxaparin
F: Warfarin
(6 months)

N/A

Rahmati, 2017

59 (F)

Yes

H/H/H/H

Diverticulosis

Fusobacterium

Yes

F. nucleatum

Yes

I: Amp/sul followed

by ertapenem
F: Ceftriaxone, metro

followed by

Amp/clav
(16 weeks)

Enoxaparin
(8 weeks)

(4 months)

Cavernous transformation

of PV

Near- complete resolution

Zheng, 2016

73 (M)

Yes

N/N/N/N

Presumed lower GI

F. nucleatum

N/A

I: Cefepime
F: Clindamycin
(N/A)

I: Enoxaparin
F: Warfarin
(N/A)

N/A

Akhrass, 2015

32 (M)

Yes

N/N/N/N

Acute perforated appendicitis

F. necrophorum

N/A

I: Pipera/tazo, Vanc

followed by

Pipera/tazo, metro
F: Clindamycin
( 6 weeks)

I: Heparin
F: Warfarin
( 6 weeks)

Venography during hospitalization

Cavernous transformation of PV

N/A

Depetrillo, 2014

63 (M)

Yes

N/H/H/H

Unknown

F. nucleatum

N/A

I: Pipera/tazo
F: Ertapenem
(4 weeks)

Warfarin
(N/A)

N/A

Depetrillo, 2014

53 (M)

Yes

N/H/H/H

Unknown

F. necrophorum

N/A

Ertapenem
(4 weeks)

Warfarin
(N/A)

N/A

Buelow, 2013

65 (M)

Yes

N/A

Diverticulitis

Negative

Yes

Fusobacterium

Yes

I: Cipro, metro
F: Ceftriaxone, metro
(N/A)

I: Heparin

F: Enoxaparin
(N/A)

N/A

Kröll, 2012

34 (M)

Yes

N/A

Oropharyngeal

Negative

Yes

F. necrophorum

Yes

Ceftriaxone, metro
(N/A)

Was given but not specified
(N/A)

Done but not specified
(1 year)

Complete resolution

Shahani, 2011

34 (M)

Yes

N/N/N/N

Pancreatitis

PV, SMV, SV

Negative

Yes

F. necrophorum

Yes

I: Vanc, meropenem

followed by

tigecycline,

meropenem
F: Tigecycline
(4 weeks)

None
(N/A)

(2 months)

Cavernous transformation of the PV and formation of collaterals

Complete resolution

Hamidi, 2008

23 (M)

Yes

H/N/H/H

Unknown

PV, SMV

F. necrophorum, E. coli,

S. intermedius

N/A

Amox/clav
(10 days)

I: LMWH
F: Fluindione
(6 months)

Recanalization of PV, mesenteric cavernoma

N/A

Hamidi, 2008

41 (M)

Yes

H/H/NA/H

Unknown

PV, SMV

F. necrophorum

N/A

N/A; patient left against medical advice

Serrano, 2007

21 (F)

Yes

N/A

Unknown

Supra-hepatic vein

F. necrophorum

Yes

F. necrophorum

Yes

I: Levo, metro,

imipenem

F: Amox/clav
(4 weeks)

Acenocoumarol
(N/A)

N/A

Le Roux, 2006

43 (M)

Yes

N/N/N/NA

Pancreatic prosthesis

F. nucleatum

Yes

Negative

Yes

I: Ceftriaxone,

ofloxacin
F: Amox/clav, metro
(10 months)

Heparin
( during hospitalization)

Persistence of prehepatic vein thrombosis

Complete resolution

Redford, 2005

53 (M)

Yes

H/H/H/H

Presumed subclinical appendicitis

F. necrophorum

N/A

I: Cefuroxime,

metro followed by

benzylpenicillin,

metro
F: Clindamycin
(6 weeks)

I: LMWH
F: Warfarin
( 3 months)

US on day 12

Complete resolution

N/A

Schweigart, 2005

67 (M)

Yes

N/A

Oropharyngeal

F. nucleatum

N/A

Clindamycin
(N/A)

Warfarin
(N/A)

N/A

El Braks, 2004

71 (F)

Yes

N/H/H/H

Oropharyngeal

SMV, PV

F. nucleatum

Yes

N/A

I: Pipera/tazo
F: ofloxacin
(5 weeks)

I: Heparin
F: Fluindione
( 9 months)

CT (1 week)

Partial resolution

N/A

Verna, 2004

56 (M)

Yes

H/H/H/H

Colon (ulcerative colitis)

F. nucleatum

N/A

Clindamycin
(2 weeks)

None
(N/A)

(6 months)

Persistent thrombosis

N/A

Clarke, 2003

19 (F)

Yes

N/H/H/H

Oropharyngeal

PV, SMV

F. necrophorum

Yes

F. necrophorum

Yes

Benzyl-penicillin, metro, cipro
(6.5 weeks)

Warfarin
(N/A)

(7 weeks)

Portal HTN, and formation of collaterals

Partial resolution

Etienne, 2001

68 (M)

Yes

No (leukopenia)

H/H/NA/NA

Diverticulosis

F. nucleatum

Yes

N/A

I: Cefotaxime, metro
F: Metro
( 5 weeks)

None
(N/A)

US (5 weeks)

N/A

Complete resolution

Soo, 1999

31 (M)

Yes

H/H/H/H

Presumed GI

SMV, PV

F. necrophorum

N/A

I: Cipro, metro,

penicillin
F: Amox/clav, metro
(6 weeks)

I: Heparin
F: Warfarin
( 6 months)

MRI

(4 months)

Resolving infarction

N/A

Bultink, 1999

23 (M)

Yes

H/H/H/N

Presumed Oropharyngeal

F. nucleatum

N/A

I: Imipenem
F: IV penicillin
(6 weeks)

Heparin
(Few days then
   stopped due to

varices)

N/A

Persistent thrombosis

N/A

Abbreviations: CT; computerized tomography, F; female, H: High, HV; hepatic vein, IJV; internal jugular vein, IMV; inferior mesenteric vein, IVC; inferior vena cava, M; male, MRI; magnetic resonance imaging, MRCP; Magnetic resonance cholangiopancreatography, N/A; not available, , s/p; status post, PV; portal vein, PVT; portal vein thrombosis, SMV; superior mesenteric veins, SV; splenic vein, US; ultrasound.

Table: Table 1: Demographic, clinical manifestation, diagnostic work-up and management for patients with GI variant of Lemierre Syndrome

Disclosures:

Fouad Jaber indicated no relevant financial relationships.

Saqr Alsakarneh indicated no relevant financial relationships.

Tala Alsharaeh indicated no relevant financial relationships.

Azizullah Beran indicated no relevant financial relationships.

John Campbell indicated no relevant financial relationships.

Mohammad Jaber indicated no relevant financial relationships.

Ahmed-Jordan Salahat indicated no relevant financial relationships.

Natalie Wilson indicated no relevant financial relationships.

Yazan Abboud indicated no relevant financial relationships.

Mohammad Almeqdadi indicated no relevant financial relationships.

Laith Numan indicated no relevant financial relationships.

Nikki duong indicated no relevant financial relationships.

Hassan Ghoz indicated no relevant financial relationships.

John Helzberg indicated no relevant financial relationships.

Wendell Clarkston indicated no relevant financial relationships.