Download PDF
CC BY-NC-ND 4.0 · Int Arch Otorhinolaryngol 2021; 25(04): e633-e640
DOI: 10.1055/s-0040-1721337
Update Article

Pediatric Patient with Lemierre Syndrome of the External Jugular Vein: Case Report and Literature Review

Yehuda Schwarz 
1   Department of Otolaryngology Head and Neck Surgery, Samson Assuta Ashdod University Hospital, Faculty of Health Sciences, Ben Gurion University of the Negev, Ashdod, Israel
,
Nadeem Habashi 
1   Department of Otolaryngology Head and Neck Surgery, Samson Assuta Ashdod University Hospital, Faculty of Health Sciences, Ben Gurion University of the Negev, Ashdod, Israel
,
Noa Rosenfeld-Yehoshua 
2   Department of Pediatric Intensive Care Unit, Samson Assuta Ashdod University Hospital, Faculty of Health Sciences, Ben Gurion University of the Negev, Ashdod, Israel
,
Eugene Soikher 
3   Department of Radiology, Samson Assuta Ashdod University Hospital, Faculty of Health Sciences, Ben Gurion University of the Negev, Ashdod, Israel
,
Tal Marom 
1   Department of Otolaryngology Head and Neck Surgery, Samson Assuta Ashdod University Hospital, Faculty of Health Sciences, Ben Gurion University of the Negev, Ashdod, Israel
,
Sharon Ovnat Tamir 
1   Department of Otolaryngology Head and Neck Surgery, Samson Assuta Ashdod University Hospital, Faculty of Health Sciences, Ben Gurion University of the Negev, Ashdod, Israel
› Author Affiliations
› Further Information
   Permissions and Reprints

Abstract

Introduction Lemierre syndrome (LS) involving the external jugular vein (EJV) is rare, and only a few cases have been reported in the literature.

Objectives To report a case of LS involving the external jugular vein as well as to make a review of the literature regarding both diagnosis and management strategies.

Data Synthesis We describe a case of LS involving the EJV and review the literature of previously published articles to search for additional cases. A PubMed, Embase, Scopus, and Web of science-based search was performed to determine the scope of coverage in well-reported articles in English. Twenty-one papers were retrieved and documented for age, incidence, pathogen, presenting symptoms, imaging, treatment, and outcome, which were noted for each of these cases. In our literature review of 21 papers, there were 16 patients (61%) in their 2nd and 3rd decades of life. Lemierre syndrome was shown to affect females and males equally. The presenting symptoms were a sore throat and fever. Treatment requires intravenous antibiotics, and there is no consensus regarding treatment with anticoagulation.

Conclusions The present case report and review of the literature emphasize the importance of history taking as well as physical examination in what seems to be a case of simple tonsillitis.

Keywords

Lemierre syndrome - external jugular vein - pharyngitis - thrombosis


Publication History

Received: 16 April 2020

Accepted: 02 October 2020

Article published online:
19 February 2021

© 2021. Fundação Otorrinolaringologia. This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonDerivative-NonCommercial License, permitting copying and reproduction so long as the original work is given appropriate credit. Contents may not be used for commecial purposes, or adapted, remixed, transformed or built upon. (https://creativecommons.org/licenses/by-nc-nd/4.0/)

Thieme Revinter Publicações Ltda.
Rua do Matoso 170, Rio de Janeiro, RJ, CEP 20270-135, Brazil

 

  • References

  • 1 Lemierre A. On certain septicemias due to anaerobic organisms. Lancet 1936; 1: 701-703
    CrossrefGoogle Scholar
  • 2 Gupta V, Tuli A, Choudhry R, Agarwal S, Mangal A. Facial vein draining into external jugular vein in humans: its variations, phylogenetic retention and clinical relevance. Surg Radiol Anat 2003; 25 (01) 36-41
    CrossrefPubMedGoogle Scholar
  • 3 Dool H, Soetekouw R, van Zanten M, Grooters E. Lemierre's syndrome: three cases and a review. Eur Arch Otorhinolaryngol 2005; 262 (08) 651-654
    CrossrefPubMedGoogle Scholar
  • 4 Ridgway JM, Parikh DA, Wright R. et al. Lemierre syndrome: a pediatric case series and review of literature. Am J Otolaryngol 2010; 31 (01) 38-45
    CrossrefPubMedGoogle Scholar
  • 5 Rebelo J, Nayan S, Choong K, Fulford M, Chan A, Sommer DD. To anticoagulate? Controversy in the management of thrombotic complications of head & neck infections. Int J Pediatr Otorhinolaryngol 2016; 88: 129-135
    CrossrefPubMedGoogle Scholar
  • 6 Cupit-Link MC, Nageswara Rao A, Warad DM, Rodriguez V. Lemierre Syndrome: A Retrospective Study of the Role of Anticoagulation and Thrombosis Outcomes. Acta Haematol 2017; 137 (02) 59-65
    CrossrefPubMedGoogle Scholar
  • 7 Lu MD, Vasavada Z, Tanner C. Lemierre syndrome following oropharyngeal infection: a case series. J Am Board Fam Med 2009; 22 (01) 79-83
    CrossrefPubMedGoogle Scholar
  • 8 Screaton NJ, Ravenel JG, Lehner PJ, Heitzman ER, Flower CD. Lemierre syndrome: forgotten but not extinct--report of four cases. Radiology 1999; 213 (02) 369-374
    CrossrefPubMedGoogle Scholar
  • 9 Puymirat E, Biais M, Camou F, Lefèvre J, Guisset O, Gabinski C. A Lemierre syndrome variant caused by Staphylococcus aureus. Am J Emerg Med 2008; 26 (03) 380.e5-380.e7
    CrossrefPubMedGoogle Scholar
  • 10 Judd O, Medcalf M, Malik T. Case 2: An unusual presentation of an unusual condition (Case Presentation). Acta Paediatr 2008; 97 (06) 694
    CrossrefPubMedGoogle Scholar
  • 11 Çıvgın E, Toprak U, Parlak S, Özer H. Fissuration of vertebral artery mycotic aneurysm due to Lemierre syndrome. Diagn Interv Imaging 2018; 99 (01) 43-45
    CrossrefPubMedGoogle Scholar
  • 12 David R. Hansberry, Michael D'Angelo, Arpan V.Prabhu, at el. Lemierre's syndrome: Acute oropharyngeal infection leading to septic thrombophlebitis of the internal jugular vein with pulmonary septic emboli. Interdiscip Neurosurg 2020; 19: 100573
    CrossrefGoogle Scholar
  • 13 Kidambi TD, Lee C, Kohlwes RJ. MRSA-Associated Lemierre's Syndrome in an Intravenous Drug User. J Gen Intern Med 2015; 30 (12) 1886-1887
    CrossrefPubMedGoogle Scholar
  • 14 Williams MD, Kerber CA, Tergin HF. Unusual presentation of Lemierre's syndrome due to Fusobacterium nucleatum. J Clin Microbiol 2003; 41 (07) 3445-3448
    CrossrefPubMedGoogle Scholar
  • 15 Young BJ, Steele RW. A teenager with sore throat and neck pain. Clin Pediatr (Phila) 2011; 50 (04) 355-356
    CrossrefPubMedGoogle Scholar
  • 16 Karkos PD, Asrani S, Karkos CD. et al. Lemierre's syndrome: A systematic review. Laryngoscope 2009; 119 (08) 1552-1559
    CrossrefPubMedGoogle Scholar
  • 17 Schwartz HC, Nguyen DC. Postanginal septicaemia with external jugular venous thrombosis: case report. Br J Oral Maxillofac Surg 1999; 37 (02) 144-146
    CrossrefPubMedGoogle Scholar
  • 18 Takiguchi J, Sakamoto H, Inoue N. Lemierre's Syndrome Variant with External Jugular Vein Thrombosis. Intern Med 2017; 56 (10) 1271-1272
    CrossrefPubMedGoogle Scholar
  • 19 Hagiya H, Haruki Y, Otsuka F. Lemierre syndrome involving external jugular vein. Acute Med Surg 2014; 2 (01) 64-68
    CrossrefPubMedGoogle Scholar
  • 20 Shibasaki Warabi Y, Yoshikawa H, Idezuka J, Yamazaki M, Onishi Y. Cerebral infarctions and brain abscess due to Lemierre syndrome. Intern Med 2005; 44 (06) 653-656
    CrossrefPubMedGoogle Scholar
  • 21 Gunatilake SS, Yapa LG, Gallala M, Gamlath R, Rodrigo C, Wimalaratna H. Lemierre's syndrome secondary to community-acquired methicillin-resistant Staphylococcus aureus infection presenting with cardiac tamponade, a rare disease with a life-threatening presentation: a case report. Int J Emerg Med 2014; 7: 39
    CrossrefPubMedGoogle Scholar
  • 22 Hagelskjaer Kristensen L, Prag J. Lemierre's syndrome and other disseminated Fusobacterium necrophorum infections in Denmark: a prospective epidemiological and clinical survey. Eur J Clin Microbiol Infect Dis 2008; 27 (09) 779-789
    CrossrefPubMedGoogle Scholar
  • 23 Abe H, Kisara A, Yagishita Y. A case of Lemierre syndrome. Intensive Care Unit 1998; 22: 281-285
    Google Scholar
  • 24 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 (06) 458-465
    CrossrefPubMedGoogle Scholar
  • 25 Herek PA, Lewis T, Bailitz JM. An unusual case of Lemierre's syndrome due to methicillin-resistant Staphylococcus aureus. J Emerg Med 2010; 39 (05) 644-646
    CrossrefPubMedGoogle Scholar
  • 26 Righini CA, Karkas A, Tourniaire R. et al. Lemierre syndrome: study of 11 cases and literature review. Head Neck 2014; 36 (07) 1044-1051
    CrossrefPubMedGoogle Scholar
  • 27 Lee S, Rutar T, Velez FG, Rosenbaum AL. Lemierre's syndrome with fourth nerve palsy. J AAPOS 2009; 13 (01) 107-108
    CrossrefPubMedGoogle Scholar
  • 28 Kuppalli K, Livorsi D, Talati NJ, Osborn M. Lemierre's syndrome due to Fusobacterium necrophorum. Lancet Infect Dis 2012; 12 (10) 808-815
    CrossrefPubMedGoogle Scholar
  • 29 Johannesen KM, Bodtger U. Lemierre's syndrome: current perspectives on diagnosis and management. Infect Drug Resist 2016; 9: 221-227
    CrossrefPubMedGoogle Scholar