Pediatr Infect Vaccine.  2016 Aug;23(2):143-148. 10.14776/piv.2016.23.2.143.

A Case of Lemierre Syndrome Manifests with Persistent Fever and Neck Stiffness Following Acute Oropharyngeal Infection

• Lee Y
• Hong YS
• Kim SS

• KMID: 2353395
• DOI: http://doi.org/10.14776/piv.2016.23.2.143

Abstract

Lemierre syndrome is a rare disease involving multiple organs affected by septic emboli following oropharyngeal infection. After the introduction of penicillin in the 1940s, it became a "forgotten" disease. However, due to the development of diagnostic image modalities including neck computed tomography (CT) scan, the number of published reports of Lemierre syndrome and diagnosis has been increasing since the 1990s. In this report, we describe a case of Lemierre syndrome, following oropharyngeal infection in a 16-year-old patient, who manifested with persistent fever and neck stiffness. Neck ultrasonography confirmed thrombus formation in the right internal jugular vein without definite evidence of septic emboli to other organs. After the three-week-long antibiotics therapy was completed, the thrombus in the right internal jugular vein finally disappeared.

Keyword

Lemierre syndrome; Thrombophlebitis

MeSH Terms

Adolescent
Anti-Bacterial Agents
Diagnosis
Fever*
Humans
Jugular Veins
Lemierre Syndrome*
Neck*
Penicillins
Rare Diseases
Thrombophlebitis
Thrombosis
Ultrasonography
Anti-Bacterial Agents
Penicillins

Figure

• 

  Fig. 1 Right jugular vein thrombus (arrow) was observed from the intracranial portion to the fourth C-spine level with patent blood flow.

• 

  Fig. 2 Shrinkage of thrombosis in the right jugular vein after parenteral antibiotics therapy. (A) A 3.0×0.36 cm elongated solid mass was observed. (B) Intraluminal thrombus was measured approximately 2.21×0.32 cm. (C) Intraluminal thrombus was measured approximately 1.03×0.16 cm. (D) Follow up ultrasonography was done on sixth day after discharge. The resolution of thrombus was confirmed.

Reference

1. Lemierre A. On certain septicaemia due to anaerobic organisms. Lancet. 1936; 227:701–703.

2. Chirinos JA, Lichtstein DM, Garcia J, Tamariz LJ. The evolution of Lemierre syndrome: report of 2 cases and review of the literature. Medicine (Baltimore). 2002; 81:458–465.

3. Ramirez S, Hild TG, Rudolph CN, Sty JR, Kehl SC, Havens P, et al. Increased diagnosis of Lemierre syndrome and other Fusobacterium necrophorum infections at a children's hospital. Pediatrics. 2003; 112:e380–e385.

4. Bae YA, Lee IJ, Kim HB, Hong MS, Lee K, Lee Y, et al. Lemierre syndrome: a case report. J Korean Radiol Soc. 2006; 54:7–10.

5. Kim EJ, Park JH, Kim CH, Park JY, Jung TH, Cha SI. A case of Lemierre syndrome associated with septic pulmonary emboli. Tuberc Respir Dis. 2005; 58:73–77.

6. Venglarcik J. Lemierre's syndrome. Pediatr Infect Dis J. 2003; 22:921–923.

7. Screaton NJ, Ravenel JG, Lehner PJ, Heitzman ER, Flower CD. Lemierre syndrome: forgotten but not extinct--report of four cases. Radiology. 1999; 213:369–374.

8. Karkos PD, Asrani S, Karkos CD, Leong SC, Theochari EG, Alexopoulou TD, et al. Lemierre's syndrome: a systematic review. Laryngoscope. 2009; 119:1552–1559.

9. Hagelskjaer LH, Prag J, Malczynski J, Kristensen JH. Incidence and clinical epidemiology of necrobacillosis, including Lemierre's syndrome, in Denmark 1990–1995. Eur J Clin Microbiol Infect Dis. 1998; 17:561–565.

10. Batty A, Wren MW, Gal M. Fusobacterium necrophorum as the cause of recurrent sore throat: comparison of isolates from persistent sore throat syndrome and Lemierre's disease. J Infect. 2005; 51:299–306.

11. Centor RM, Atkinson TP, Ratliff AE, Xiao L, Crabb DM, Estrada CA, et al. The clinical presentation of Fusobacterium-positive and streptococcal-positive pharyngitis in a university health clinic: a cross-sectional study. Ann Intern Med. 2015; 162:241–247.

12. Golpe R, Marin B, Alonso M. Lemierre's syndrome (necrobacillosis). Postgrad Med J. 1999; 75:141–144.

13. Forrester LJ, Campbell BJ, Berg JN, Barrett JT. Aggregation of platelets by Fusobacterium necrophorum. J Clin Microbiol. 1985; 22:245–249.

14. Goldenberg NA, Knapp-Clevenger R, Hays T, Manco-Johnson MJ. Lemierre's and Lemierre's-like syndromes in children: survival and thromboembolic outcomes. Pediatrics. 2005; 116:e543–e548.

15. Riordan T. Human infection with Fusobacterium necrophorum (Necrobacillosis), with a focus on Lemierre's syndrome. Clin Microbiol Rev. 2007; 20:622–659.

16. Kuppalli K, Livorsi D, Talati NJ, Osborn M. Lemierre's syndrome due to Fusobacterium necrophorum. Lancet Infect Dis. 2012; 12:808–815.

17. Bondy P, Grant T. Lemierre's syndrome: what are the roles for anticoagulation and long-term antibiotic therapy? Ann Otol Rhinol Laryngol. 2008; 117:679–683.

18. Syed MI, Baring D, Addidle M, Murray C, Adams C. Lemierre syndrome: two cases and a review. Laryngoscope. 2007; 117:1605–1610.

19. Hagelskjaer KL, Prag J. Human necrobacillosis, with emphasis on Lemierre's syndrome. Clin Infect Dis. 2000; 31:524–532.