Ludwig?s angina is a rare disorder characterized by a rapidly expanding and potentially life-threatening infection of the submandibular and sublingual spaces. It usually occurs in young adults with dental infections. Symptoms include severe neck pain and swelling, fever, and dysphagia. Respiratory distress and stridor suggest an impending airway catastrophe. Although most patients recover without complications, Ludwig?s angina can be fatal, as illustrated in this case.

Case Presentation
A 47-year-old woman presented to the emergency department with a toothache in a right molar as well as facial swelling and pain of 4 days? duration. She was drooling and had dysphagia, difficulty speaking, and difficulty opening her mouth. Her medical history included hypertension, cocaine use, and poor oral hygiene.

Significant physical examination findings were: temperature, 100.4?F; blood pressure, 108/63 mm Hg; heart rate, 120 beats/min; respiratory rate, 30 breaths/ min. Erythema and swelling were noted on the right lower jaw and the left side of her face; these areas felt warm, tense, and taut, but with no crepitus. Her tongue was protruding beyond the lips (Figure 1), and the lower right second molar was necrotic. The patient had no stridor.

Laboratory tests revealed leukocytosis, with a white blood cell count of 27.8 x 10⁹/L. Computed tomography (CT) demonstrated swelling with inflammatory changes and air within the soft tissues along the right and left mandible (Figure 2). Urine drug screening was positive for cocaine. Blood cultures were negative.

The emergency department diagnosis was Ludwig?s angina originating from the necrotic right second molar. The patient was electively nasally intubated using a fiberoptic laryngoscope and was then transferred to the intensive care unit (ICU).

At admission, a regimen of 3 g intravenous (IV) ampicillin sodium/sodium sulbactam (Unasyn) every 6 hours was started. Clindamycin (Cleocin), 900 mg IV, followed by 600 mg IV every 8 hours, and methylprednisolone (A-Methapred, Solu-Medrol), 60 mg IV every 8 hours, were added 2 days later.

The patient underwent surgical procedures on hospital days 5 and 11, during which copious amounts of foul-smelling pus were drained. The first procedure also included extraction of 2 carious molars from the right mandible. No bacteria were found in a sample taken during the first surgical procedure, but a culture obtained from the submental drain a few days later grew Pseudomonas aeruginosa. The patient?s condition continued to deteriorate, and she died on hospital day 14.

Autopsy revealed an abscess in the floor of the mouth and the upper right neck area. Postmortem blood cultures and culture samples from the neck abscess and lungs grew P aeruginosa.

Discussion
Ludwig?s angina was first described by the German physician Wilhelm Friedrich von Ludwig in 1836. At that time, the condition was almost always fatal.¹ With the advent of contrast-enhanced CT, which has allowed earlier identification and prompt antibiotic therapy, the mortality rate has significantly declined, from more than 50% to less than 10% of patients.^2,3

Ludwig?s angina is a rapidly spreading, indurated, bilateral cellulitis that begins in the floor of the mouth and involves both the submandibular and sublingual spaces.⁴ It spreads along fascial planes rather than by lymphatics and rarely involves the glandular surfaces.⁵

The primary site of infection is odontogenic in 70% to 80% of cases.⁶ The second and third molars are most frequently involved, because their roots extend below the level of the mylohyoid muscle, thus crossing both the sublingual and submandibular spaces.⁷ The majority of the patients are adults who have no significant comorbidities, but this condition has also been associated with systemic diseases, such as chronic glomerulonephritis, systemic lupus erythematosus, aplastic anemia, neutropenia, immunodeficiency (eg, HIV infection), diabetes mellitus, and hypersensitivity.⁸ Illegal IV drug use and trauma are predisposing factors.⁸

The bacteriology of Ludwig?s angina is polymicrobial. The most common organisms identified include Streptococcus, Staphylococcus, and Bacteroides species. Other microorganisms that have been isolated are gram-negative bacteria, such as Klebsiella species, Hemophilus influenzae, Proteus species, and P aeruginosa.⁹

Clinical manifestations
Pain in the floor of the mouth and anterior neck, dysphagia, odynophagia, and respiratory distress are common symptoms.¹⁰ Clinical findings include fever, tachypnea, and tachycardia, and patients may also have fetid breath. Stridor, hoarseness, respiratory distress, cyanosis, and decreased air movement are harbingers of impending upper airway compromise.

Palpation of the submental and bilateral submaxillary spaces reveals firm, nonpitting induration of the suprahyoid neck bilaterally.⁷ Inspection of the malodorous oral cavity is limited because of trismus, but a firm, raised floor of the mouth may be evident.⁷

Although abscess formation is not always associated with Ludwig?s angina, some cases will eventually evolve into an abscess. Complications of Ludwig?s angina include sepsis, pneumonia, asphyxia, empy?ema, pericarditis, mediastinitis, and pneumothorax.⁴

Diagnosis
Diagnosis is based on clinical findings, although contrast-enhanced CT can help determine the extent of the infection, especially in the presence of an abscess.⁸ Clinical examination has a low sensitivity (55%) for predicting drainable collections of pus in deep neck infections, but when combined with CT findings, the accuracy is 89%, sensitivity is 95%, and specificity is 80% for identifying a drainable collection.¹¹ Plain radiographs of the neck may show soft-tissue swelling, the presence of gas, and the extent of airway narrowing.

The differential diagnosis of Ludwig?s angina includes angioneurotic edema, lingual carcinoma, sublingual hematoma, and peritonsillar abscess.

Airway management
Because of the risk of rapid airway compromise, all patients with Ludwig?s angina should be admitted to the ICU. Death is usually the result of hypoxia or asphyxia, not overwhelming sepsis,² although sepsis was the likely cause in our patient. Airway management is the most important aspect of immediate care. Tracheostomy using local anesthesia has been considered the gold standard of airway management in patients with deep-neck infections. However, cellulitis of the neck with involvement of the tracheostomy site makes the procedure difficult.¹² A recent case series that included 17 patients with Ludwig?s angina showed that tracheal intubation with a flexible bronchoscope using topical anesthesia provided good airway management. The authors recommended tracheostomy using local anesthesia when fiberoptic intubation is not possible.¹⁰

Antibiotic therapy
Initial antibiotic therapy is targeted at gram-positive organisms and oral cavity anaerobes. Empiric therapy with IV penicillin G, clindamycin, or metro?nidazole is recommended before culture and antibiogram results are available.⁸ Some experts recommend the addition of gentamicin.⁸ Antibiotic treatment before hospital admission often results in sterile cultures. IV dexamethasone (eg, Dalalone, Decadron, Dexasone), given for 48 hours, can decrease edema and cellulitis and thus help maintain the integrity of the airway and enhance antibiotic penetration.¹³ If an abscess is present, the definitive treatment would be incision and drainage and, if applicable, removal of the abscessed tooth or teeth.

Drainage
Surgical drainage is indicated in the presence of clinical fluctuance or crepitus, or radiologic evidence of fluid collection or air in the soft tissues. A relative indication is the lack of clinical improvement within 24 hours of initiation of antibiotic therapy.¹ Removal of infected teeth facilitates the complete drainage of fluid.

Conclusion
Ludwig?s angina usually resolves without complications, but the condition can be fatal. Prompt diagnosis, appropriate airway management, aggressive IV antibiotic therapy, and close monitoring in the ICU promote good outcomes in most patients.

References
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11. Miller WD, Furst IM, Sandor GK, et al. A prospective, blinded comparison of clinical examination and computed tomography in deep neck infections. Laryngoscope. 1999;109:1873-1879.

12. Mehrotra M, Mehrotra S. Decompression of Ludwig angina under cervical block. Anesthesiology. 2002; 97: 1625-1626.

13. Ramadan HH, El Solh AA. An update on otolaryngology in critical care. Am J Respir Crit Care Med. 2004; 169:1273-1277.