Ludwig’s angina, anterior neck abscess and cavernous venous thrombosis following third molar extraction

Abstract

Ludwig’s angina (LA) is a rapidly progressing cellulitis that develops after oral infections or molar extractions, typically causing fever and chills and rarely progressing to trismus and cyanosis. It is associated with risk factors including diabetes mellitus, heavy alcohol use, oral malignancy, poor dentition or an immunocompromised state. This case report reviews a previously healthy patient with no appreciable risk factors presenting with LA following a third molar extraction, complicated by extensive anterior deep neck space abscesses, cavernous venous thrombosis and Lemierre’s Syndrome. Our case demonstrates the importance of early intervention when suspecting LA to prevent life-threatening complications and death.

Background

Third molar ‘wisdom tooth’ extraction surgery is the most common ambulatory procedure done by oral and maxillofacial surgeons.¹ While modern surgery techniques aim to reduce complications, there are nonetheless unintended consequences that may result from an extraction, which usually include pain, swelling, ecchymosis, trismus, infection and nerve damage. A rare but serious complication of third molar extraction is Ludwig’s angina (LA) secondary to infection of the lower molar teeth.^{2 3}

LA is an aggressive, rapidly progressive inflammation and cellulitis involving the submandibular, sublingual and/or submental spaces.⁴ This potentially life-threatening polymicrobial odontogenic infection typically presents with fever, chills and malaise, which can progress to trismus, drooling, stridor and cyanosis.⁵ Due to the immediate risk of airway compromise, aggressive treatment with airway preservation and intravenous antibiotics are the mainstay of therapy.⁶ If a drainable collection is observed on imaging, needle aspiration or incision and drainage (I&D) may be performed. Common risk factors for LA are screened for prior to any invasive dental procedure and include diabetes, previous oral malignancy, dental caries, alcoholism, HIV or any other immunocompromised state, and recent exposure to methicillin-resistant Staphylococcus aureus (MRSA).⁷

While LA following third molar extraction is a rare complication, further infection spread along fascial planes of the neck and subsequent abscess formation is even less common.⁸ The exact incidence of these complications from extraction is not well known, but LA is known to be caused by third molar extraction seeding odontogenic bacteria into the surrounding tissue in around 90% of cases.⁷

Case presentation

A previously healthy woman in her early 20s presented to the emergency department with neck swelling, pain, trismus, dysphagia and purulent drainage consistent with LA following three wisdom tooth extractions 11 days prior. She was initially evaluated in the outpatient setting for these symptoms 7 days after the procedure and failed outpatient management with amoxicillin/clavulanate, metronidazole, chlorhexidine, dexamethasone and cyclobenzaprine. While oral and maxillofacial surgery prescribed this regimen in an outpatient setting, double anaerobic coverage is an uncommon practice that is generally not recommended. She had a rapid progression of her symptoms and presented to the emergency department 11 days postprocedure, with trismus (maximum interincisal opening of <10 mm), severe submental and bilateral submandibular swelling and erythema that extended below the level of the hyoid, as well as profound neck tenderness. At the time of presentation, the inferior border of the mandible was non-palpable, and her oral cavity was unable to be evaluated due to severe swelling. She was found to be afebrile at 36.8°C but tachycardic to 120 bpm and had leukocytosis of 34x10ˆ9/L with a left shift, meeting systemic inflammatory response criteria for sepsis secondary to oral infection.^{9 10} CT neck with contrast showed extensive left anterior neck abscess which appeared to be originating from the masticator space (figure 1) and two small lesions in the bilateral upper lung lobes. The patient underwent I&D with surgical drains placed. She was started on vancomycin, ceftriaxone and metronidazole and was intubated postprocedure due to swelling, after which the patient self-extubated. Abscess culture grew Streptococcus constellatus and Prevotella spp while blood cultures were negative.

Figure 1.

The first CT neck with contrast obtained for our patient, demonstrating a large anterior neck abscess.

Two days postoperative, CT thorax with contrast was done to investigate the upper lung lesions, which revealed several bilateral pulmonary septic emboli (figure 2). Transthoracic echocardiogram was negative for endocarditis and septal defects. Due to worsening pain and swelling, a CTA head and neck and CT maxillofacial was done, which showed worsening temporal and masseter abscess, cavernous sinus thrombosis (figure 3), left internal jugular vein septic phlebitis (Lemierre’s syndrome) and narrowing of the cavernous segments of the internal carotid arteries (ICA) bilaterally. She underwent repeat I&D with debridement; abscess culture grew S. constellatus and Candida albicans. Antibiotics were continued in addition to fluconazole for candidiasis and heparin in the setting of cavernous venous thrombosis.

Figure 2.

CT thorax with contrast which demonstrates numerous septic emboli in the bilateral lungs.

Figure 3.

CT head with contrast demonstrating cavernous venous thrombosis.

In the following days, the patient’s symptoms improved and leucocytosis downtrended. Due to previous evidence of ICA narrowing, she received a diagnostic cerebral angiogram which revealed right ICA stenosis of 60%–70% and left ICA stenosis of 70%–80%, with the likely aetiology deemed to be related to endovascular infection (figure 4). As part of the workup for her complications, the patient was screened for underlying risk factors. She was found to be HIV negative, is a non-smoker and is an occasional social drinker. She does not have any history of dental caries or other recent infections. The patient had no other appreciable risk factors or predisposing factors.

Figure 4.

Cerebral angiogram of the left interior carotid artery, demonstrating significant stenosis.

Treatment

The patient underwent several I&D procedures with debridement and drain placement. Culture-directed therapies with antibiotics and antifungals were used for her infections and heparin was administered in the setting of cavernous venous thrombosis. The patient received 10 hyperbaric oxygen (HBO) treatments in preparation for anterior cervical skin graft. Due to several heparin therapy interruptions for HBO sessions, the patient was switched from heparin to apixaban for anticoagulation. On completion of HBO therapy, she underwent uncomplicated split thickness skin grafting of the anterior neck. The graft and donor site healed adequately, and the patient was discharged home on apixaban for a duration of 3 months due to provoked cavernous venous thrombosis.

Outcome and follow-up

The graft and donor site healed adequately, and the patient was discharged home. On outpatient follow-up 2 weeks later, the patient had appropriate healing of the graft and donor sites, with no postoperative concerns or complications. Approximately 7 months later, when contacted for consent in regard to this case report, the patient reported complete wound healing and return to her baseline health, and continued ability to complete her daily activities without restriction.

Discussion

LA is a rare and possibly fatal complication of an infection involving the lower molar teeth, with second and third molar extractions being the most likely cause.^{2 4} All patients should be screened for comorbid conditions prior to receiving dental care, as predisposing factors such as diabetes mellitus or immunosuppression may contribute to developing LA.^{4 6 7} Evaluating LA is dependent on whether or not the patient’s airway is secure, as asphyxiation (oxygen deprivation), is the most common cause of death for this pathology.⁸ Our patient’s presentation in the emergency department was consistent with typical symptoms of LA, without signs of airway obstruction. Due to our patient’s confirmed airway patency, we first obtained a CT head with intravenous contrast, which is the imaging modality of choice and assesses for thickening of soft tissue, fluid collections and muscular oedema.

Our patient had a complicated clinical course including extensive anterior deep neck space abscesses, cavernous venous thrombosis and Lemierre’s syndrome. Cavernous venous thrombosis is a rare but serious health complication that involves cerebral vein occlusions, occurring in about 2 individuals per 100 000. The pathophysiology of this complication is from the oral cavity infection spreading in a retrograde fashion. Other LA complications may include aspiration pneumonia, carotid arterial rupture, sheath abscess, necrotising fasciitis, mediastinitis and pericarditis.^6–8 Although abscess formation is rare in LA, it was present in this patient. Common organisms that may cause abscesses in LA include Streptococcus viridans and oral anaerobes such as Actinomyces spp and Fusobacterium spp.⁶

Treatment for LA is guided by multiple factors including airway patency, immune status and surgical intervention.⁶ Immune status is pertinent for empiric antibiotic treatment; our patient was immunocompetent but received broad antimicrobial coverage due to the purulent nature of her infection. Additionally, empirical vancomycin therapy was recommended for our patient by infectious disease specialists due to the acuity of her presentation as well as increased prevalence of MRSA in the local flora. For our patient, oral maxillofacial surgery was involved early in care as an abscess was present, and the patient underwent two incisions and drainages of bilateral abscess of the deep neck spaces. LA is quite rare, along with further complications of abscess formation and cavernous venous thrombosis, which makes our case unique especially in a young woman without any appreciable risk factors. Our case demonstrates the importance of early intervention when suspecting LA, as early diagnosis and treatment are crucial in preventing life-threatening complications and death.

Learning points.

• Known risk factors for Ludwig’s angina include diabetes mellitus, heavy alcohol use, oral malignancy, poor dentition and an immunocompromised state.

• While patients with known risk factors are more susceptible to oral infections such as Ludwig’s Angina, our case demonstrates that healthy patients with no predisposing factors may still suffer devastating sequelae of Ludwig’s angina.

• Early diagnosis and intervention is of paramount importance when suspecting Ludwig’s angina to prevent life-threatening complications and death.

Ethics statements

Patient consent for publication

Consent obtained directly from patient(s).