Patients with Lemierre syndrome have a high risk of new thromboembolic complications, clinical sequelae and death: an analysis of 712 cases

L Valerio; F Zane; C Sacco; S Granziera; T Nicoletti; M Russo; G Corsi; K Holm; M-A Hotz; C Righini; P D Karkos; S H Mahmoudpour; N Kucher; P Verhamme; M Di Nisio; R M Centor; S V Konstantinides; A Pecci; S Barco

doi:10.1111/joim.13114

Patients with Lemierre syndrome have a high risk of new thromboembolic complications, clinical sequelae and death: an analysis of 712 cases

J Intern Med. 2021 Mar;289(3):325-339. doi: 10.1111/joim.13114. Epub 2020 Jun 18.

Authors

L Valerio¹, F Zane², C Sacco³, S Granziera⁴, T Nicoletti⁵, M Russo¹, G Corsi⁶, K Holm⁷, M-A Hotz⁸, C Righini⁹, P D Karkos¹⁰, S H Mahmoudpour^{1

11}, N Kucher¹², P Verhamme¹³, M Di Nisio¹⁴, R M Centor¹⁵, S V Konstantinides^{1

16}, A Pecci¹⁷, S Barco^{1

12}

Affiliations

¹ From the, Center for Thrombosis and Hemostasis, University Medical Center Mainz, Mainz, Germany.
² Department of General Medicine, Hospital of Sondrio, Sondrio, Italy.
³ Thrombosis and Hemostasis Center, Humanitas Research Hospital and Humanitas University, Rozzano, Italy.
⁴ Department of Physical and Rehabilitation Medicine, "Villa Salus" Hospital, Mestre, Italy.
⁵ Institute of Neurology, Catholic University of the Sacred Heart and Institute of Neurology, Fondazione Policlinico A. Gemelli IRCCS, Rome, Italy.
⁶ Department of Emergency Medicine, San Giovanni Calibita Fatebenefratelli Hospital, AFAR, Rome, Italy.
⁷ Department of Clinical Sciences, Division of Infection Medicine, Lund University, Lund, Sweden.
⁸ Department of ENT, Head and Neck Surgery, Inselspital, University of Bern, Bern, Switzerland.
⁹ Department of ENT, Head and Neck Surgery, University Hospital of Grenoble, Grenoble, France.
¹⁰ Department of Otolaryngology-Head and Neck Surgery, AHEPA Hospital, Aristotle University of Thessaloniki, Thessaloniki, Greece.
¹¹ Institute for Medical Biostatistics, Epidemiology, and Informatics (IMBEI), Department of Biometry and Bioinformatics, University Medical Center Mainz, Mainz, Germany.
¹² Clinic of Angiology, University Hospital Zurich, Zurich, Switzerland.
¹³ Department of Vascular Medicine and Hemostasis, University Hospitals Leuven, Leuven, Belgium.
¹⁴ Department of Medicine and Ageing Sciences, University G. D'Annunzio of Chieti-Pescara, Chieti, Italy.
¹⁵ Huntsville Regional Medical Campus, University of Alabama Birmingham School of Medicine, Birmingham, AL, USA.
¹⁶ Department of Cardiology, Democritus University of Thrace, Alexandroupolis, Greece.
¹⁷ Department of Internal Medicine, IRCCS Policlinico San Matteo Foundation and University of Pavia, Pavia, Italy.

PMID: 32445216
DOI: 10.1111/joim.13114

Abstract

Background: Lemierre syndrome is characterized by head/neck vein thrombosis and septic embolism usually complicating an acute oropharyngeal bacterial infection in adolescents and young adults. We described the course of Lemierre syndrome in the contemporary era.

Methods: In our individual-level analysis of 712 patients (2000-2017), we included cases described as Lemierre syndrome if these criteria were met: (i) primary site of bacterial infection in the head/neck; (ii) objectively confirmed local thrombotic complications or septic embolism. The study outcomes were new or recurrent venous thromboembolism or peripheral septic lesions, major bleeding, all-cause death and clinical sequelae.

Results: The median age was 21 (Q1-Q3: 17-33) years, and 295 (41%) were female. At diagnosis, acute thrombosis of head/neck veins was detected in 597 (84%) patients, septic embolism in 582 (82%) and both in 468 (80%). After diagnosis and during in-hospital follow-up, new venous thromboembolism occurred in 34 (5.2%, 95% CI 3.8-7.2%) patients, new peripheral septic lesions became evident in 76 (11.7%; 9.4-14.3%). The rate of either was lower in patients who received anticoagulation (OR: 0.59; 0.36-0.94), higher in those with initial intracranial involvement (OR: 2.35; 1.45-3.80). Major bleeding occurred in 19 patients (2.9%; 1.9-4.5%), and 26 died (4.0%; 2.7-5.8%). Clinical sequelae were reported in 65 (10.4%, 8.2-13.0%) individuals, often consisting of cranial nerve palsy (n = 24) and orthopaedic limitations (n = 19).

Conclusions: Patients with Lemierre syndrome were characterized by a substantial risk of new thromboembolic complications and death. This risk was higher in the presence of initial intracranial involvement. One-tenth of survivors suffered major clinical sequelae.

Keywords: Fusobacterium necrophorum; Lemierre syndrome; anticoagulation; pulmonary embolism; septic embolism; venous thromboembolism.

Publication types

Research Support, Non-U.S. Gov't
Review

MeSH terms

Adolescent
Adult
Disease Progression
Female
Humans
Lemierre Syndrome / complications*
Lemierre Syndrome / mortality
Male
Thromboembolism / etiology*
Thromboembolism / mortality
Venous Thrombosis / etiology*
Venous Thrombosis / mortality