A Rare Twist of the Forgotten Disease: A Case of Fusobacterium necrophorum Sepsis with Portomesenteric Thrombosis and a Review of the Literature

Abstract

Abdominal variants of Lemierre’s syndrome presenting with pylephlebitis are rare, and the role of anticoagulation in treatment is controversial. We hereby report a case of pylephlebitis secondary to F. necrophorum bacteremia in a 57-year-old female originating from bacterial translocation secondary to colitis, who developed a favorable outcome with prompt treatment with antibiotics and anticoagulation. We also perform a literature review on similar cases in the literature and discuss management options of this rare but potentially fatal complication.

Pylephlebitis Caused by Fusobacterium nucleatum in a Septuagenarian Healthy Caucasian Male: Atypical Presentation of Lemierre’s Syndrome

“…Lemierre's syndrome is a specific form of septic thrombophlebitis, which includes venous thrombosis associated with local bacterial infection occurring at several anatomic locations [ 11 ]. Current literature describes a variant of Lemierre's syndrome involving an older demographic who present with an initial infection and thrombosis of the portal vein [ 8 , 12 ]. The pathogenesis of the thrombus formation remains the same; however, the strain of Fusobacterium involved in the portal vein thrombosis differs from that of a typical case of Lemierre's syndrome.…”

“…Typically, Fusobacterium necrophorum is involved, but in the atypical cases, the responsible pathogen points to Fusobacterium nucleatum [ 12 ]. In light of the recent report by Lazar et al, describing several cases of portomesenteric thrombosis due to Fusobacterium necrophorum infection [ 8 ], atypical Lemierre's syndrome may be attributed to Fusobacterium spp. infection.…”

“…Evidence from a case report dating back to 2014 mentioned two cases involving portal vein thrombosis in context of Fusobacterium infections. The report mentioned that while Fusobacterium is known to induce platelet aggregation, thrombotic events outside of the head and neck are rare [ 8 , 14 ]. When they do occur, they are mainly seen in elderly patients and mortality can approach 25% [ 8 , 14 ].…”

“…The report mentioned that while Fusobacterium is known to induce platelet aggregation, thrombotic events outside of the head and neck are rare [ 8 , 14 ]. When they do occur, they are mainly seen in elderly patients and mortality can approach 25% [ 8 , 14 ].…”

“…Fusobacterium sepsis, more commonly associated with thrombophlebitis of the internal jugular vein, also known as Lemierre's syndrome, is a rare cause of pylephlebitis or other forms of thrombophlebitis outside of the head-neck syndrome, especially in the gastrointestinal system. These conditions are sometimes referred to as atypical Lemierre's syndrome [ 7 , 8 ]. In this report, we present the case of a Fusobacterium -associated thrombophlebitis of the portal vein in an otherwise healthy Caucasian male.…”