Investigation of the Association between Recreational Scuba Diving and

a Case of Acute Lemierre’s Syndrome: Will it Recur?

DIVYA A. PANDYA, GLEN E. SUTHERLAND, RACHEL SLACK, MICHAEL F. BLACKARD,

DAVID DROLLER, PAUL AGTARAP

Department of Graduate Medical Education, Internal Medicine Residency Program,

Broward Health Medical Center,

1600 S Andrews Ave, Fort Lauderdale, FL 33316,

UNITED STATES OF AMERICA

Abstract: - Lemierre’s Syndrome (LS) is a septic thrombophlebitis of the internal jugular vein following acute

pharyngitis commonly caused by anaerobic bacteria. We postulate a rare source of infection in a young male

never before cited in the literature, scuba diving. We discuss the patient’s risk factors which we postulate likely

caused an altered innate immunity, and when combined with superimposed barotrauma, likely provoked the

infection. We then consider the possibility of disease recurrence and make certain recommendations for

prevention. Additionally, we highlight the importance of early diagnosis in potentially critical diseases such as

LS.

Key-Words: - Lemierre’s Syndrome, septic thrombophlebitis, allergic rhinosinusitis, barotrauma, diving, altered

immunity.

Received: April 18, 2022. Revised: January 11, 2023. Accepted: February 16, 2023. Published: March 14, 2023.

1 Background

Lemierre’s Syndrome (LS) has been identified to

be a complication of oropharyngeal infection

classically presenting as sore throat and persistent

fevers progressing to septic thrombophlebitis of the

vascular region of the lateral neck, [1], [2].

In a recent systemic review of 84 case reports,

the most common source of infection was found to

be the pharynx and tonsils with sources of lungs,

larynx, teeth, eyes, and the mastoid process being

less common, [3]. The most commonly identified

causative agent, Fusobacterium necrophorum, is

part of the normal flora of the human upper

respiratory tract, [3], [4]. Another recent systemic

review of 712 cases found that 84% of patients had

acute thrombosis of head/neck vasculature, and

82% had septic embolisms, [5]. LS affects young

individuals with a median age of 21 years and has a

substantial risk of major clinical sequelae, various

new thromboembolic complications, and even

death, [4], [5].

Antibiotics are the mainstay of treatment, with

carbapenems, metronidazole, and piperacillin-

tazobactam being the initial choice targeting gram-

negative bacilli and anaerobic microorganisms

common in the disease, [6]. Recommendations and

guidelines regarding systemic anticoagulation,

however, are inconsistent and have not been

investigated in clinical trials. Some systemic

reviews of cases have shown lower rates of new

venous thromboembolisms and septic peripheral

lesions in those that received anticoagulation, [5].

Here, we present a case of Lemierre's Syndrome

with a likely unique cause of infection. Although

difficult to prove without a doubt, it is likely the

patient acquired his infection from his diving

activities. A case associated with recreational scuba

diving has never prior been cited in the available

English literature. Additionally, we highlight the

importance of further thorough investigation and

testing when a diagnosis is unable to be made

initially or does not fit the clinical picture

appropriately.

2 Case Description

Our patient is a 24-year-old male who presented to

our institution as an international transfer from

Trinidad and Tobago. His medical history consisted

of a deviated septum, chronic allergic

rhinosinusitis, and recurrent sinus pain diagnosed

by ear, nose, and throat (ENT) specialists as

neuropathic in nature. Of note, the patient’s

profession is as a commercial diver. It requires him

to dive to a maximum of 70 feet underwater for at

least 60 minutes at a time.

His symptoms began while on vacation in

Barbados as a left-sided sore throat. He went

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DOI: 10.37394/23208.2023.20.4

Divya A. Pandya, Glen E. Sutherland,

Rachel Slack, Michael F. Blackard,

David Droller, Paul Agtarap

E-ISSN: 2224-2902

Volume 20, 2023

recreational scuba diving one time 6-12 hours prior

to symptom onset. After the dive, he had some self-

limited mild diarrhea. However, 4 days after

symptom onset, he had an episode of light

headedness, headaches, sweats, chills, and fever

that prompted him to go to the emergency room

(ER) upon return to Trinidad. After multiple daily

visits to the ER, he was eventually admitted to the

hospital on day 10 of his illness. A chest radiograph

(RA) showed bilateral patchy infiltrates and he was

admitted for presumed viral pneumonia with a

possible superimposed bacterial infection. Multiple

COVID-19 antigen and PCR tests were negative

and it was noted that he had received the

Sinopharm COVID vaccination. He was treated

with 3 days of azithromycin, ceftriaxone, and

oseltamivir, and due to symptomatic improvement

of his sore throat, he was subsequently discharged

on day 13 of his illness. However, 2 days later, on

day 15, he returned with worsening leukocytosis

and continued fever and chills which at this point

had lasted almost 2 weeks. He was started on

piperacillin/tazobactam and levofloxacin. Further

testing including blood cultures was negative. After

6 days of total antibiotics, his computed

tomography (CT) of the chest showed worsening

bilateral pleural effusions and cavitating pulmonary

lesions.

Fig. 1: CT chest with IV contrast of our patient on

1/22/2022. Bilateral lung cavitary nodules and

pleural effusions, likely septic pulmonary emboli,

left lower lobe consolidation with some pockets of

air may represent developing abscess.

Due to a lack of clinical improvement, and new

worsening cavitary pulmonary nodules, the patient

was internationally transferred to our institution on

day 22. CT chest with contrast showed persistent

cavitary pulmonary nodules bilaterally, small sub-

segmental septic pulmonary emboli, small bilateral

pleural effusions, and a lower left consolidation

with pockets of air concerning developing an

abscess, (Figure 1). Our final blood cultures were

negative, although this is likely due to previous

antibiotic treatment. Due to concern for LS, a

venous ultrasound doppler (US) of the upper left

extremity was completed revealing a deep vein

thrombosis (DVT) of the left internal jugular vein

(IJV), (Figure 3 A-B).

Fig. 2: CT chest with IV contrast of our patient on

1/27/2022. Overall significant improvement

inflammatory process and decreased size cavitary

nodules and pleural effusions.

A diagnosis of LS was made based on the

characteristic clinical course of the sore throat

followed by fevers accompanied by IJV thrombus

and septic pulmonary emboli. The patient was

continued on intravenous piperacillin-tazobactam

and therapeutic anticoagulation throughout his

hospitalization. He was discharged on day 32 with

6 weeks of oral metronidazole, 3 months of oral

anticoagulation, and close follow-up appointments

with the infectious disease specialist. Prior to

discharge, the patient’s repeat venous US doppler

and CT chest with contrast showed marked

improvement in the size of the DVT and septic

pulmonary emboli (Figure 2 and Figure 3 C-D).

3 Discussion

Our patient’s initial symptoms of sore throat and

fever were nonspecific and overlooked requiring

multiple ER visits. Despite eventual

hospitalization, a definitive diagnosis reflecting his

condition wasn’t established until far into his

clinical course. We would like to highlight the

importance of maintaining a wide differential and

pursuing diligent diagnostic workup when a

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DOI: 10.37394/23208.2023.20.4

Divya A. Pandya, Glen E. Sutherland,

Rachel Slack, Michael F. Blackard,

David Droller, Paul Agtarap

E-ISSN: 2224-2902

Volume 20, 2023

common presumed diagnosis doesn’t match the

clinical course. Especially in the younger patient

population, it is imperative to keep not only LS in

mind but also various other rare medical conditions

as they can be easily overlooked due to nonspecific

presenting symptoms. In many diseases such as LS,

an earlier diagnosis prevents devastating

complications.

Fig. 3: Doppler Ultrasounds of Left Internal Jugular

Vein

A-B: US Doppler on 1/25/2022 reveals a large

DVT of Left Internal Jugular Vein and superficial

partial thrombophlebitis involving the left cephalic

vein.

C-D: US Doppler on 1/31/2022 show considerable

improvement in DVT of Left Internal Jugular Vein

and minimal improvement in thrombus in left

cephalic vein.

LS can develop from the local spread of certain

oropharyngeal pathogens, [1]. It can be associated

with regional thrombophlebitis in an otherwise

normal host, in patients with a history of allergic

rhinosinusitis, or with predisposing viral or

bacterial nasal-oropharyngeal infection, [1], [2].

Our patient did have a history of allergic

rhinosinusitis which we hypothesize was likely a

factor predisposing him to infection due to altered

innate immunity from chronic inflammation and

impaired clearance of sinus ostia, [9]. Among the

oropharyngeal pathogens normally found in the

human respiratory tract, Fusobacterium

necrophorum is one of the most virulent species

capable of causing opportunistic infections,

especially in the setting of altered innate immunity,

[4]. This species and various others may share

virulence factors that facilitate regional septic

thrombophlebitis. Heparinase, an exotoxin, enables

invasion without tissue destruction, [7]. Another

exotoxin, hemolysin, develops an anaerobic

environment for other anaerobic organisms such as

Fusobacterium necrophorium to grow.

Additionally, endotoxins such as Leucocidin may

play a role, by lysing neutrophils. To prevent the

propagation of our patient’s internal jugular

thrombus further into the cerebral vasculature, or

the formation of further thrombi, we initiated

therapeutic dose anticoagulation. Recall,

recommendations regarding systemic

anticoagulation are inconsistent, [5]. Regardless, on

repeat imaging, our patient had marked

improvement in the size of the IJV thrombosis and

septic pulmonary emboli. Treatment with

anticoagulation in addition to antibiotics may be

beneficial. Further clinical trials are needed to

establish guidelines.

Now, our hypothesis is that the recreational

scuba dive may have predisposed the patient to

develop LS. The offending pathogens had not been

identified in our patient’s case, likely due to the

receipt of empiric broad-spectrum antibiotics prior

to blood culture draws. However, our patient may

have acquired a virulent pathogen via a

contaminated mouthpiece or other scuba

equipment. He may also have acquired LS from

swallowing contaminated water which may explain

his self-limited diarrhea, [8].

Our patient also had anatomical anomalies that

likely predisposed him to LS. He had a surgically

uncorrected deviated septum and probable mucosal

thickening from chronic allergic rhinosinusitis

leading to sinus obstruction from blocked ostia,

subsequently preventing nasal passage clearance,

predisposing him to infection, [7]. Given our

patient’s chronic rhinosinusitis, we cannot rule out

an altered innate immunity as a predisposing factor

to the spread of virulent pathogens, [9].

Additionally, the role of barotrauma must also

be seriously considered as a contributing factor,

leading to the pathophysiology of our patient’s LS,

[10], [11], [12]. His recurrent sinus and facial pain

are likely a consequence of said barotrauma.

Barosinusitis can be explained by Boyles law,

when the pressure of a gas is decreased, the volume

of gas increases. When the diver ascends, there will

be decreased pressure causing an increase in

volume, consequently compressing the mucosa,

and thus leading to ischemia. When the diver

descends, there is increased pressure causing

decreased volume which could lead to tugging with

increased mucosal capillary congestion, leaky

capillaries, and bleeding. These results will also

lead to inflammatory thrombogenic host responses,

and possibly strip endothelial cells or cause

endothelial dysfunction causing changes in the

coagulation system. Additionally, in cases due to

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DOI: 10.37394/23208.2023.20.4

Divya A. Pandya, Glen E. Sutherland,

Rachel Slack, Michael F. Blackard,

David Droller, Paul Agtarap

E-ISSN: 2224-2902

Volume 20, 2023

such anaerobes as fusobacterium, barosinusitis may

add to the microaerophilic local anatomical

environment by causing further ischemia and

allowing the anaerobic microbes to proliferate. In

other words, the change in barometric pressure

during diving causes irritation and damage to the

mucosal lining of the paranasal sinuses by failing to

equalize intranasal pressures with ambient

environmental pressure.

Finally, diving can result in barosinusitis, and

barosinusitis is an additional risk factor with our

patient’s underlying altered anatomy, [10], [11].

These risk factors in the setting of our patient’s

altered innate immunity secondary to chronic

allergic rhinosinusitis likely contributed to the

pathophysiology of LS. Hence, we can conclude

that there would be a probable risk of recurrence of

LS with subsequent dives in our patient’s case.

Without standard-of-care treatment guidelines,

we would recommend in cases of LS, six weeks of

antibiotic treatment for endovascular infection, to

cover empirically for mixed aerobic and anaerobic

oropharyngeal flora. The antibiotics could also be

further narrowed to target likely pathogens

identified from blood cultures. Additionally, we

recommend anticoagulation be prescribed for three

months although the issue of utilizing

anticoagulation in LS to minimize embolic

phenomenon remains unresolved in the medical

literature. We also recommend follow-up imaging

such as chest CT’s and venous doppler ultrasounds

to evaluate the course of the patient’s septic

thrombi.

Our patient was made aware that diving may

have contributed to the development of LS and that

we are unsure of the risk of recurrence from

continuing diving. We made certain

recommendations that may ameliorate the risk of

recurrence. First, we recommend using

mouthpieces that are disinfected, according to the

CDC guidelines, [13]. Second, we recommend he

avoid contaminated environments and minimize

swallowing water, e.g., when feasible, wear a full-

face mask. Third, we recommend in order to

minimize sinus obstruction, he should consider

normalizing his deviated septum if surgically

amenable and perform allergy testing and

hyposensitization. Fourth, one should avoid or

postpone a dive if there is active infectious or acute

allergic rhinosinusitis, [11]. Fifth, it is imperative to

practice good dental hygiene. Sixth, one must

remain up-to-date on vaccinations against

respiratory pathogens. Seventh, a diver should

repeatedly practice maneuvers to equalize pressures

between sinuses and ambient environmental

pressure. These include Valsalva (pinching nostrils

and gently blowing), Frenzel (pinching nostrils

closed and attempting to make a ‘k’ sound), and

Toynbee (pinching nostrils and swallowing or

yawning), [14]. Eighth, we should consider if there

is a role for chemoprophylaxis, e.g., PrEP with

antibacterial rinses or medications.

4 Conclusion

This is an interesting case of LS demonstrating a

unique cause of infection. No other cases of LS

associated with diving were found in the English

scientific literature. Our patient likely developed

LS due to a combination of circumstances. He

suffered from recurrent barotrauma and mucosal

injury causing blocked ostia from his long history

of dives. His chronic allergic rhinosinusitis also led

to altered innate immunity. Subsequently, he was

exposed to contaminated scuba equipment and

likely swallowed contaminated water duringhis

recreational dive. Therefore, these combined

circumstances were likely the “perfect storm” that

led to his upper respiratory tract infection

predisposing him to LS. The risk of developing

recurrent LS will remain until he is able to alter

these circumstances. Therefore, if our hypothesis is

correct, LS may recur. Due to its rarity, the optimal

approach to prevent LS is unknown. Here, we

provide a foundation for an approach as we await

further corroborating evidence.

Clinical Message:

Further clinical trials and research are needed to

make guidelines not only regarding the role of

anticoagulation but also to study the possibility of

recurrence of LS due to diving and the methods of

prevention.

Consent:

Informed consent was obtained from the patient to

publish case details, test results, and images.

Competing Interests:

No competing interests to disclose.

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Divya A. Pandya, Glen E. Sutherland,

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David Droller, Paul Agtarap

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Volume 20, 2023

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DOI: 10.37394/23208.2023.20.4

Divya A. Pandya, Glen E. Sutherland,

Rachel Slack, Michael F. Blackard,

David Droller, Paul Agtarap

E-ISSN: 2224-2902

Volume 20, 2023

Contribution of Individual Authors to the

Creation of a Scientific Article (Ghostwriting

Policy)

The authors equally contributed in the present

research, at all stages from the formulation of the

problem to the final findings and solution.

Sources of Funding for Research Presented in a

Scientific Article or Scientific Article Itself

No funding was received for conducting this study.

Conflict of Interest

The authors have no conflicts of interest to declare

that are relevant to the content of this article.

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