European Annals of Otorhinolaryngology, Head and Neck diseases 133 (2016) 123–124

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Case report

Atypical Lemierre syndrome

M. Risoud ∗ , G. Mortuaire , D. Chevalier , B. Rysman
Service d’ORL et chirurgie cervico-faciale, hôpital Claude-Huriez, centre hospitalier régional universitaire de Lille, rue Michel-Polonovski, 59037 Lille cedex,
France

a r t i c l e i n f o a b s t r a c t

Keywords: Introduction: Lemierre syndrome is a complication of oropharyngeal infection and consists of a combina-
Lemierre syndrome tion of internal jugular vein septic thrombophlebitis with septicaemia and distant septic emboli (mainly
Facial vein thrombosis in the lungs). We describe an atypical case with facial vein and anterior jugular vein thrombophlebitis.
Head and neck cellulitis
Case summary: A 34-year-old woman attended the emergency room with tonsillitis, left head and neck
cellulitis, left facial vein thrombosis and lung abscesses. A diagnosis of atypical Lemierre syndrome
was proposed and the patient was treated surgically (neck incision and tonsillectomy) and medically
(antibiotics, hyperbaric oxygen therapy and anticoagulation) allowing cure without sequelae.
Discussion: Lemierre syndrome, a rare but serious complication requiring immediate treatment, should
be investigated (by blood cultures and chest CT scan) in the presence of neck vein thrombosis complicating
oropharyngeal infection.
© 2015 Elsevier Masson SAS. All rights reserved.

1. Introduction The laboratory assessment, comprising serum electrolytes, clot-

Lemierre syndrome was described by André Lemierre in 1936
[1] and corresponds to the presence of internal jugular vein septic
thrombophlebitis with septicaemia and distant septic emboli (usu-
ally lung abscesses) complicating oropharyngeal infection [2,3]. The
incidence of this severe syndrome, although rare, appears to be
slightly increasing [2].
We report a case of atypical Lemierre syndrome with septic
thrombophlebitis of the facial vein draining into the anterior jugu-
lar vein.
2. Case report
A 34-year-old woman, with no history apart from active smok-
ing, was referred to the emergency room by her general practitioner
due to the appearance of an inflammatory swelling of the left cheek
and neck associated with fever and trismus present for several
hours. These clinical features followed a 10-day episode of tonsil-
litis treated with amoxicillin for the previous 2 days.
Physical examination revealed features of left head and neck cel-
lulitis with hard, tender, inflammatory swelling of the cheek and
neck, associated with fever of 38.8 ◦ C, tachypnoea, trismus with
mouth opening reduced to 1 cm and odynophagia. Intraoral exam-
ination revealed good dental status and left tonsillitis.
∗ Corresponding author. Tel.: +33 3 20446526; fax: +33 3 20444688.
E-mail address: michael.risoud@gmail.com (M. Risoud).
ting assessment, complete blood count and CRP as well as blood
cultures, revealed an intense inflammatory syndrome with eleva-
tion of CRP to 153 mg/L and leukocytosis of 23,000/mm3 .
Dental panoramic radiography was normal. Contrast-enhanced
chest and head and neck CT scan injected (Fig. 1) revealed throm-
bosis of the left facial vein and left anterior jugular vein with
infiltration of the adjacent fat and fascia without a clearly defined
collection. Chest CT sections revealed bilateral pleural effusion with
lung abscesses.
Surgery was indicated in view of the rapidly extensive nature
of the cellulitis associated with venous thrombosis. Neck incision
and drainage as well as left tonsillectomy were performed. Empir-
ical antibiotic therapy with ceftriaxone and metronidazole was
initiated. The patient was then transferred to the intensive care
unit and remained intubated for 3 days with hyperbaric oxygen
therapy.
The first two blood cultures identified the pathogen responsible
(Streptococcus constellatus) and antibiotic therapy was changed to
amoxicillin-clavulanate. The patient also received curative doses
of anticoagulant therapy, initially with calcium hepari, followed by
vitamin K antagonist (fluindione).
The clinical course was favourable and the patient was dis-
charged from hospital after two weeks with continuation of
vitamin K antagonist therapy for 3 months with a target INR
between 2 and 3.
One month after discharge, no sequelae were detected on
physical examination and follow-up chest CT scan demonstrated
resolution of the lung abscesses.

http://dx.doi.org/10.1016/j.anorl.2015.12.001
1879-7296/© 2015 Elsevier Masson SAS. All rights reserved.
124 M. Risoud et al. / European Annals of Otorhinolaryngology, Head and Neck diseases 133 (2016) 123–124
Fig. 1. Head and neck (A, B, C) and chest CT scan (D, E) demonstrating thrombosis of the left facial vein (A, B) and left anterior jugular vein (C) (arrows) as well as bilateral
pleural effusion with excavated pulmonary condensation suggestive of lung abscesses (stars) (D, E).
3. Discussion
Lemierre syndrome is a rare, but serious disease. Atypical forms
of this syndrome must be recognised. The pathogen responsible in
the case reported here (Streptococcus constellatus) has been previ-
ously isolated in other cases of Lemierre syndrome [3]. However,
the micro-organism most commonly identified is Fusobacterium
necrophorum [2–4].
The present case corresponds to atypical Lemierre syndrome
(in terms of the vessels involved) secondary to cellulitis aris-
ing from the tonsils. Cases of atypical Lemierre syndrome have
rarely been reported in the literature. One case of facial vein
septic thrombophlebitis has been described [5]. Another case of
F. necrophorum facial vein thrombophlebitis has been reported,
but without lung abscess [6]. Cases involving the external jugu-
lar vein and superior ophthalmic vein have also been reported
[7,8].
Treatment of atypical Lemierre syndrome, as for typical
Lemierre syndrome, comprises combination medical therapy
sometimes combined with surgery. The place of anticoagulation
remains controversial.
4. Conclusion
The presence of venous thrombosis in the neck in a context of
oropharyngeal infection should raise the suspicion of Lemierre syn-
drome and constitutes an indication for chest and head and neck
CT scan and blood cultures, even when the thrombosis does not
involve the internal jugular vein.
Disclosure of interest
The authors declare that they have no competing interest.
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