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\begin{document}

\title{A case of blindness caused by Lemierre's syndrome}



\author[1]{Tensei Suzuki}%
\author[1]{Mituaki Kojima}%
\author[1]{Raira Nakamoto}%
\author[1]{Keiichi Kuriyama}%
\author[1]{Shu Tanizawa}%
\author[1]{Yuzuru Mochida}%
\author[1]{Yuri Asakura}%
\author[1]{Ayaka Shibano}%
\author[1]{Tmohisa Shoko}%
\affil[1]{Tokyo Women's Medical University Adachi Medical Center}%


\vspace{-1em}



  
  \date{May 27, 2024}


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\textbf{Introduction}

Lemierre's syndrome is a systemic septic embolism caused by
thrombophlebitis of the internal jugular vein and presenting with
various distantly infected abscesses. Various diagnostic criteria exist,
but there is no standardized definition. Lemierre's syndrome is
generally believed to be caused by infection with anaerobic bacteria
around the oropharynx. Delayed diagnosis can lead to dyspnea and deep
neck infections due to acute airway constriction, which can lead to
fatal outcomes. Ophthalmologic complications of Lemierre's syndrome are
relatively rare. In this study, we report our experience with a patient
with Lemierre's syndrome who lost her sight.

\textbf{Case}

\emph{Patient:} A 36-year-old woman.

\emph{Chief complaints:} Fever, posterior cervical pain, facial
swelling, right visual acuity loss

\emph{Medical history:} No major diseases, including immune disorders.

\emph{Family history:} Nothing in particular.

\emph{Lifestyle history:} Nonsmoker and occasional drinker. The patient
was not on any oral medications.

\emph{History of present illness:} The patient developed a fever 1 week
before the hospital visit. Two days later, she developed difficulty in
swallowing and trismus and sore throat. By the following day, she had
facial swelling and posterior cervical pain. She kept monitoring her
condition at home. However, the symptoms did not improve, and she began
having difficulty in moving, which prompted an ambulance request 1 week
later and the patient's being transported to our hospital.

\emph{Physical findings at the hospital visit:} Height, 155 cm; body
weight, 55 kg; clear consciousness; body temperature, 38.0\selectlanguage{ngerman}°C; heart
rate, 110 bpm (regular); blood pressure, 98/76 mmHg;
SpO\textsubscript{2}, 95\% (room air); respiratory rate, 27 breaths per
minute; no chills and shivering; no night sweats; right-dominant facial
swelling; examination of the oral cavity and pharynx proved difficult
due to trismus; swelling and tenderness noted in the right cervical
area; no stiff neck; no chest pain; clear pulmonary sounds; no clear
abnormalities in the cardiac sound; flat and soft abdomen; no
spontaneous pain and tenderness; and no limb swelling.

\emph{Abnormal neurological findings (cranial nerves):} II, right visual
acuity loss; III/IV/VI, right eye midline immobilization, no oculomotor
disorder in the left eye; V, no left/right differences in facial
sensation; VII, trismus/difficulty in opening the right eye; VIII, no
left/right differences in hearing; IX/X, difficulty in swallowing; XI,
no weakness or left/right differences in the sternocleidomastoid and
trapezius muscles; XII, the tongue could not extend beyond dentition
because of trismus.

\emph{Laboratory findings at the hospital visit:} Arterial blood gas
analysis (room air) showed pH, 7.409; PaCO\textsubscript{2}, 38.9 Torr;
PaO\textsubscript{2}, 80.2Torr; and bicarbonate, 24.1 mmol/L. She had no
respiratory failure. Blood work revealed a white blood cell count of
21,900/\selectlanguage{greek}µ\selectlanguage{english}L, platelet count of 1.8\selectlanguage{ngerman}×~10\textsuperscript{4} \selectlanguage{greek}μ\selectlanguage{english}L, C-reactive
protein level of 26.73 mg/dL, quantitative fibrinogen level of 680
mg/dL, Fibrin degradation product level of 4.8 \selectlanguage{greek}µ\selectlanguage{english}g/dL, and D-dimer level
of 1.8 \selectlanguage{greek}µ\selectlanguage{english}g/dL, which indicated an elevated inflammatory response,
thrombocytopenia, and high D-dimer level. The acute stage score of
disseminated intravascular coagulation (DIC) was 4 points. Therefore,
she was diagnosed with DIC.

\emph{Images at the hospital visit:} The contrast-enhanced computed
tomography (CT) showing a low-density area in the C2-6 prevertebral
muscles, and a poorly enhanced area in the right internal jugular vein.
(Fig.1.2) There are also Multiple ground-glass nodules are in both
lungs, predominantly on the pleural side.(Fig. 3)

The patient was diagnosed with Lemierre's syndrome, based on the images
indicating retropharyngeal abscess, thrombophlebitis, and septic
pulmonary embolism.

After admission, the patient was managed in the intensive care unit.

At the time of admission, trismus was observed, and CT showed airway
stenosis. Securing the airway was necessary. Therefore, the patient
underwent emergency tracheotomy in the primary care room, after
undergoing a platelet transfusion.

Local drainage of the retropharyngeal abscess was attempted, but it was
impossible to visualize and treat the locality of the pharynx because of
trismus; therefore, conservative treatment with antibiotics was
administered. Meropenem 1 g IV every 8 hours and vancomycin 1 g IV every
12 hours were the empiric antimicrobial agents. Anticoagulant therapy
was not started on admission because the patient was in a state of DIC
at the time of admission. The patient was diagnosed with orbital-apex
syndrome due to the spread of inflammation from the retropharyngeal
abscess.

Treatments with antimicrobial agents gradually improved the inflammatory
parameters, facial swelling, and posterior cervical pain. Blood culture
sampled upon admission revealed \emph{Fusobacterium necrophorum} .
Therefore, the antimicrobial treatments were de-escalated to
ampicillin/sulbactam 3 g IV every 6 hours on Day 6 after admission,
based on the susceptibility results. Anticoagulant therapy was also
started with systemic administration of heparin. Heparin was switched to
a direct-acting oral anticoagulant on Day 11, as the patient had no
exacerbation in her general condition.

On Day 35, antimicrobial treatments were de-escalated to amoxicillin 500
mg orally 3 times daily. Antimicrobial treatments were terminated on Day
42, after confirming that the abscess had disappeared completely on the
follow-up CT scan.

However, even after the abscess disappeared, the patient's reduced
visual acuity and oculomotor disorder did not improve. The patient was
discharged with these symptoms remaining.

\textbf{Discussion}

In our patient, the period from the preceding infection to the
appearance of Lemierre's syndrome symptoms was short; thus, we
understand that an extremely intense condition manifested in our
patient.

\emph{Fusobacterium necrophorum} , a gram-negative anaerobic bacteria,
is the causative agent of Lemierre's syndrome in most cases. The same
bacterium was identified in our patient. The most common organ affected
by septic embolism is the lungs, followed by joints such as hip joints,
knees, and shoulders; skin and soft tissue; and the
endocardium.\textsuperscript{1,2}In a systematic review in 2020, Dasari
et al.\textsuperscript{3} found only 27 cases of ophthalmologic
complications with Lemierre's disease reported between 2009 and
2019.\textsuperscript{4} Among these 27 cases, the most common
ophthalmologic complications were cranial nerve III/IV/VI palsy and
oculomotor disorder due to external ophthalmoplegia. Abducens nerve
palsy was noted in 12 (44.44\%) cases. Cavernous sinus thrombosis was
observed in 19 (70.37\%) cases. The next most common symptoms were
blepharoptosis (9 cases, 33.33\%) and visual impairment (8 cases,
29.63\%). Of the three patients with visual impairment, the condition of
two patients recovered to some extent, but that of the remaining one
patient did not.\textsuperscript{3}

Ophthalmologic complications of Lemierre syndrome may be caused by
inflammation spreading from the cavernous sinus to the nerves and
muscles. The cavernous sinus receives blood from various veins such as
the superior orbital vein, intracranial vein, and parietal sinus. These
extensive connections cause retrograde septic embolism from the internal
jugular vein, resulting in cavernous sinus
thrombosis.\textsuperscript{3,4}

\textbf{Conclusion}

We describe a patient who lost her vision because of Lemierre's
syndrome. Ophthalmologic complications of Lemierre's syndrome are rare,
and very few cases have been reported. Clinicians need to recognize that
it is a serious condition that can lead to blindness in some cases.



\textbf{Conflict of interest}

Authors declare no Conflict of Interests for this article.

\textbf{Acknowledgements}

I would like to thank all the staff of the Department of Emergency
Medicine for their support in the treatment of patients. The abstract of
this paper was presented in 2022 at the 50\textsuperscript{th} Annual
Meeting of the Japanese Association for Acute Medicine.

\textbf{Ethics Statement}

\textbf{Approval of the research protocol: N/A}

\textbf{Informed consent:} The patient provided informed consent for
treatment and for the report to be published.

\textbf{Registry and the registration no. of the study/trial} : N/A.

\textbf{Animal studies:} N/A.

\textbf{Conflict of interest:} Authors declare no Conflict of Interests
for this article.

\textbf{Data Sharing and Data Accessibility} : Not applicable.

\textbf{References}

1. Miller B, Khalifa Y, Feldon SE, Friedman DI. Lemierre syndrome
causing bilateral cavernous sinus thrombosis. J. Neuroophthalmol. 2012;
32: 341--4.

2. Gutzeit A, Roos JE, Portocarrero-Fa\selectlanguage{english}h B, et al. Differential diagnosis
of Lemierre's syndrome in a patient with acute paresis of the abducens
and oculomotor nerves. Korean J. Ophthalmol. 2013; 27: 219--23.

3. Dasari SP, Jha P. A systematic review of Lemierre's Syndrome with a
focus on ophthalmologic complications. Cureus. 2020; 12: e9326.

4. Ebright JR, Pace MT, Niazi AF. Septic thrombosis of the cavernous
sinuses. Arch. Intern. Med. 2001; 161: 2671--6.

\textbf{Figure Legends}

Fig. 1 The cervical contrast-enhanced CT image showing a low-density
area in the C2-6 prevertebral muscles. This is denoted by the orange
circle.

CT, computed tomography

Fig. 2 A poorly enhanced area in the right internal jugular vein. This
is denoted by the orange circle.

Fig.3 Multiple ground-glass nodules are in both lungs, predominantly on
the pleural side. This is denoted by the orange circle.

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