Journal of Infection and Public Health 13 (2020) 1360–1362

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Journal of Infection and Public Health

journal homepage: http://www.elsevier.com/locate/jiph

Case Report

A 4-year-old boy with Lemierre’s syndrome caused by

methicillin-sensitive Staphylococcus aureus
Tahir Hameed a,b,c,∗ , Manal Bawazeer a,b,c , Nora Alfattoh a,b,c , Sami Alanazi a,b
a
Department of Pediatrics, King Abdullah Specialized Children’s Hospital, King Abdulaziz Medical City, Ministry of National Guard – Health Affairs, Riyadh,
Saudi Arabia
b
King Saud bin Abdulaziz University for Health Sciences, Riyadh, Saudi Arabia
c
King Abdullah International Medical Research Center, Riyadh, Saudi Arabia

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

Article history: A pre-school aged boy presented to the Pediatric Emergency Department with a high grade fever and
Received 30 March 2020 neck pain and stiffness. Blood culture was positive for methicillin-sensitive Staphylococcus aureus (MSSA)
Received in revised form 12 May 2020 and Doppler ultrasound of the neck revealed partial thrombosis of the left internal jugular vein. He
Accepted 17 May 2020
was diagnosed with Lemierre’s syndrome (LS) and treated with a prolonged course of antibiotics and
anticoagulation. After discharge home, he was followed in the outpatient clinics and had a full recovery.
Keywords:
This case report will highlight the presentation of LS and will briefly review the microbiology of this
Lemierre’s syndrome
condition.
Septic thrombophlebitis
© 2020 The Authors. Published by Elsevier Ltd on behalf of King Saud Bin Abdulaziz University for
Fusobacterium
Staphylococcus aureus
Health Sciences. This is an open access article under the CC BY-NC-ND license (http://creativecommons.
org/licenses/by-nc-nd/4.0/).

Introduction spectrum antibiotics followed by pathogen-specific regimens once

In 1936, the French physician Dr. Andre Lemierre described the
condition named after him. He described 20 cases of suppurative
thrombophlebitis of the internal jugular vein (IJV) [1]. Lemierre’s
syndrome (LS) is an uncommon condition that classically starts
with an oropharyngeal infection (like tonsillitis, retropharyngeal
abscess or dental infection) which is complicated by suppurative
thrombophlebitis of the IJV and subsequent septic emboli to the
lungs or other organs [2,3]. The highest incidence is in the second
decade of life [3].
Fusobacterium necrophorum, an anaerobic gram-negative bacil-
lus, is the most common cause of LS [4]. In a review of the literature,
approximately 82% of the cases of LS were caused by this bac-
terium. However several other organisms have been reported to
cause this condition, including other anaerobes as well as gram-
positive bacteria like streptococcal and staphylococcal species [5].
Some authors limit the diagnosis of LS only for those conditions
in which there is thrombophlebitis of the IJV caused by anaer-
obic organisms (mainly F. necrophorum) following a preceding
oropharyngeal infection [6]. The initial management of LS is broad-
∗ Corresponding author at: Department of Pediatrics – Mail Code 1940, King
Abdullah Specialized Children’s Hospital, King Abdulaziz Medical City – Central
Region; King Saud bin Abdulaziz University for Health Sciences, Ministry of National
Guard – Health Affairs, PO Box 22490, Riyadh 11426, Saudi Arabia.
E-mail address: hameedta@ngha.med.sa (T. Hameed).
culture data become available. Though most patients respond to
antibiotics alone, in selected cases there is a need for surgical inter-
vention (for example for uncontrolled local infection or metastatic
spread like a large lung abscess). The role of anticoagulant therapy
in LS is controversial; it may be indicated when there is significant
extension of the thrombus [7–9].
Although LS was once considered a rare disease, internationally
there has been an increase in the number of reported cases. It is not
known if the increase in cases is due to an actual increase in the inci-
dence of the disease or other factors such as an increase in antibiotic
resistance, a decrease in tonsillectomies or a decrease in the pre-
scription of antibiotics [10,11]. While Fusobacterium species have
been historically the predominant bacteria causing this condition,
we report a pediatric patient with methicillin-sensitive Staphylo-
coccus aureus (MSSA) as the causative agent in LS.
Case summary
A previously healthy 4-year-old Saudi boy was assessed in the
Pediatric Emergency Department (ED) with an acute history of
fever, neck pain, and abdominal pain. He had visited the ED two
days prior and was diagnosed with a viral upper respiratory tract
infection. Further history also revealed trauma to the back of his
head in the preceding days; however no abrasion or signs of skin or
soft tissue infection were seen. On initial examination, the child
looked unwell; he exhibited grunting and was in pain. He was
highly febrile on presentation with tachycardia and tachypnea. The

https://doi.org/10.1016/j.jiph.2020.05.013
1876-0341/© 2020 The Authors. Published by Elsevier Ltd on behalf of King Saud Bin Abdulaziz University for Health Sciences. This is an open access article under the CC
BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
 T. Hameed et al. / Journal of Infection and Public Health 13 (2020) 1360–1362
Fig. 1. Doppler ultrasound of the neck showing partial thrombosis of the left internal
jugular vein.
initial diagnosis in the ED was compensated septic shock to rule out
meningitis. After appropriate resuscitation, the child was started
on Ceftriaxone and Vancomycin. Brain computed tomography (CT)
was done which was normal, and a lateral neck X-ray revealed slight
thickening of the prevertebral soft tissue. The child required admis-
sion to the pediatric intensive care unit (PICU). In the PICU, the
child had increasing left-sided neck pain and swelling as well as a
left 6th cranial nerve palsy. Neck CT with contrast was done and
revealed no definite retropharyngeal abscess. However there was a
left jugular vein filling defect. As this was suggestive of thrombosis,
Doppler ultrasound of the neck was performed and revealed partial
thrombosis of the left IJV (Fig. 1).
In addition, CT of the brain with contrast was done and showed
no evidence of dural vein thrombosis. The blood culture was pos-
itive for MSSA while the cerebrospinal fluid culture was negative.
A diagnosis of Lemierre’s syndrome was made with the findings
of IJV thrombosis and MSSA bacteremia and sepsis. After ini-
tial broad-spectrum antibiotic coverage, he received IV floxacillin
(150 mg/kg/day divided Q6H).
About 3 weeks into the child’s hospitalization, a new swelling
was noted on the left side of his neck. Neck ultrasound showed sus-
picion of a pseudoaneurysm arising from the left vertebral artery.
CT angiogram of the neck revealed a left paravertebral inflamma-
tory mass at the level of C2 and left vertebral artery thrombosis. The
child subsequently underwent a procedure in which coiling of the
left vertebral artery was done at the orifice of the pseudoaneurysm.
Anticoagulation was started.
The child had an otherwise uncomplicated hospital course, and
completed an 8 week course of IV antibiotics. He had a normal
neurological examination prior to discharge. Upon discharge, the
child was prescribed high dose Cephalexin (100 mg/kg/day divided
Q8H) which he received for approximately 7 weeks. The child
was also to complete 6 months of treatment with aspirin. Follow-
up was given with multiple services including General Pediatrics,
Neurology, Ophthalmology, and Infectious Diseases. On outpatient
follow-up, the child continued to show complete recovery and had
normal development for his age.
Discussion
While there have been few reported case reports of Lemierre’s
syndrome (LS) in Saudi Arabia, we believe this condition is under-
recognized and thus under-reported. We present this case report
1361
of a pre-school aged Saudi boy with LS to raise awareness of this
condition as well as to highlight the increase in cases of S. aureus-
induced LS.
LS often presents with sore throat, fever, and a swollen/tender
neck [5]. Some patients may appear septic and have absence of
throat symptoms [12]. Thus a high index of suspicion is needed
to identify and diagnose this condition. Though Fusobacterium spp.
are most commonly implicated with LS, reports suggest that other
pathogens are associated with LS or Lemierre-like syndrome. In
a recent review, Van Hoeke et al. reviewed all reported cases of
S. aureus-induced Lemierre-like syndrome. There were 25 case
reports; interestingly all these reports were since 2002 and many
of the cases were in young children. Out of the pediatric cases, 6 out
of 9 were caused by methicillin-resistant S. aureus (MRSA). Given
that S. aureus (including MRSA) is an emerging cause of LS, ini-
tial antibiotic choices should include anaerobic and MRSA coverage
[13].
With the changing microbiology of LS, the clinician should
be aware of increasingly virulent and antibiotic-resistant organ-
isms. There are reported cases of MSSA causing LS. Pathogenic
strains of MSSA are increasingly Panton-Valentine leucocidin (PVL)
producers. PVL appears to have pathogenic synergism with alpha-
hemolysin released by staphylococci, giving it the ability to lyse
leukocytes and erythrocytes causing immune evasion and bacte-
rial replication. This shared feature of PVL-producing S. aureus and F.
necrophorum may explain the development of LS by both organisms
[14].
Conclusion
Though uncommon, Lemierre’s syndrome can be a fatal dis-
ease that may present with a meningitis or sepsis-like picture. This
infection-induced suppurative thrombophlebitis of the IJV with
septic emboli may be unreported and should not be “the forgot-
ten disease” [15]. Clinicians should have a high index of suspicion
for this condition in a child presenting with sore throat, neck pain,
fever, and a toxic appearance. While Fusobacterium species are con-
sidered to be the most common causes of LS, other bacteria can
cause this condition such as MSSA isolated in our patient. More case
studies are needed to show if there is in fact changing epidemiology
of the causative agents for LS in the pediatric age group.
Funding
No funding sources.
Competing interests
None declared.
Ethical approval
Not required.
Acknowledgements
The authors would like to thank Dr. Amna Kashgari for providing
the radiological image for this patient.
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