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J Stomatol Oral Maxillofac Surg xxx (2020) xxx–xxx

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A 6-month-old infant girl presents with neck stiffness

N. Cramer *, D. Azhdam
Division of Pediatric Emergency Medicine, AOB 2400, UPMC Children’s Hospital of Pediatrics, 4401, Penn Avenue, 15224 Pittsburgh, PA, United States

1. Case report

A 6-month-old female presented to the pediatric emergency

department with five days of fever, vomiting, diarrhea, and
shoulder stiffness. Vital signs were remarkable for fever and
tachycardia. There was restricted active movement but normal
passive range of motion of her head and neck. The patient was
admitted and laboratory studies revealed a leukocytosis of
24.5  10E + 09/L, bandemia of 15%, hemoglobin of 8.8 g/dL
(88 g/L), and elevated CRP of 18.9 mg/dL (0.189 g/L). Two days
later, a chest radiograph revealed left lower lobe opacities with a
moderate pleural effusion. Shortly thereafter, the patient devel-
oped apnea necessitating intubation. In the pediatric intensive care
unit, the patient was noted to have anterior neck fullness. Soft
tissue ultrasound revealed a 6.7  3.3  4.8 cm abscess (Fig. 1)
which CT confirmed extended from the floor of the mouth to the

Fig. 2. Sagittal CT scan revealing deep neck space abscess with extension into the
mediastinum.

mediastinum (Fig. 2). There was associated airway compression,

right internal jugular vein thrombosis, and left upper lobe
pneumonia with tree-in-bud formation in the right upper lobe.
What is your diagnosis?

Fig. 1. Neck ultrasound revealing a large midline deep neck abscess that is
6.7  3.3  4.8 cm in size.

Abbreviation: LS, Lemierre syndrome.

* Corresponding author.
E-mail addresses: natan.cramer@chp.edu (N. Cramer), devora.azhdam@chp.edu
(D. Azhdam).

https://doi.org/10.1016/j.jormas.2020.03.019
2468-7855/ C 2020 Elsevier Masson SAS. All rights reserved.

Please cite this article in press as: Cramer N, Azhdam D. A 6-month-old infant girl presents with neck stiffness. J Stomatol Oral
Maxillofac Surg (2020), https://doi.org/10.1016/j.jormas.2020.03.019
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2 N. Cramer, D. Azhdam / J Stomatol Oral Maxillofac Surg xxx (2020) xxx–xxx
2. Answer
The patient was diagnosed with Lemierre syndrome (LS). The
abscess was drained, and both abscess and blood cultures grew
Streptococcus milleri.
3. Brief discussion
Our case of a 6-month-old with LS represents one of the
youngest cases reported [1]. The definition of LS includes a
complicated pharyngeal infection involving septic thrombophle-
bitis of an internal jugular vein and distant septic emboli [2]. Deep-
space neck infections spread quickly along continuous fascial
planes and abruptly cause life-threatening sequelae due to the
limited amount of room for swelling to diffuse, which contributes
to rapid airway compression. The differential diagnosis for
Lemierre syndrome includes; retropharyngeal abscess, Ludwig’s
angina, descending necrotizing mediastinitis as well as epiglottis
[3]. Imaging helps differentiates between these etiologies,
however, certain clinical clues such as tripoding for epiglottitis
and neck fullness for Ludwig’s angina can help guide diagnosis
[3]. The classic causative bacterium in LS is Fusobacterium
necrophorum, although other microbes have been implicated.
F. necrophorum infection is typically biphasic, occurring after a
resolving case of pharyngitis [2]. Conversely, the timeline of
S. milleri LS infection, as in our patient, is monophasic with
progressive worsening. Patients with LS improve with drainage
and antimicrobial therapy but the utility of anticoagulation for
associated thromboses remains controversial [4].
Please cite this article in press as: Cramer N, Azhdam D. A 6-month-old infant girl presents with neck stiffness. J Stomatol Oral
Maxillofac Surg (2020), https://doi.org/10.1016/j.jormas.2020.03.019
It was suspected that the patient had an underlying congenital
anatomic anomaly predisposing her to this devastating infection,
such as a lymphatic malformation or thyroglossal duct cyst. The
patient ultimately was found to have a thyroglossal duct cyst on a
repeat ultrasound, which was removed via Sistrunk procedure.
Thyroglossal duct cysts are rarely diagnosed acutely with life-
threatening soft tissue infectious symptoms. Typically, mildly
symptomatic midline swelling is the presenting marker for this
underlying anomaly. Ultrasound is a reasonable initial diagnostic
study and recurrence is relatively rare [5].
Disclosure of interest
The authors declare that they have no competing interest.
References
[1] Garcı́a-Salido A, Unzueta-Roch JL, Garcı́a-Teresa MÁ, Sirvent-Cerdá S, Martı́nez
de Azagra-Garde A, Casado-Flores J. Pediatric disseminated lemierre syndrome
in 2 infants: not too young for an ancient disease. Pediatr Emerg Care
2017;33(7):490–3. http://dx.doi.org/10.1097/PEC.0000000000000570.
[2] Osowicki J, Kapur S, Phuong LK, Dobson S. The long shadow of Lemierre’s
syndrome. J Infect 2017;74:S47–53. http://dx.doi.org/10.1016/s0163-
4453(17)30191-3.
[3] Li RM, Kiemeney M. Infections of the neck. Emerg Med Clin North Am
2019;37(1):95–107. http://dx.doi.org/10.1016/j.emc.2018.09.003.
[4] 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(2):59–65. http://dx.doi.org/10.1159/000452855.
[5] Al-Thani H, El-Menyar A, Sulaiti MA, et al. Presentation, management, and
outcome of thyroglossal duct cysts in adult and pediatric populations: a 14-year
single center experience. Oman Med J 2016;31(4):276–83. http://dx.doi.org/
10.5001/omj.2016.54.