20 November 2023: Articles
Unusual Case of Ludwig Angina Caused by in an Immunocompromised HIV-Positive Patient with Alcoholism and Dental Abscess
Challenging differential diagnosis, Management of emergency care, Clinical situation which can not be reproduced for ethical reasons, Rare coexistence of disease or pathologyRachel D. Truong 1ABCEF, Van Anh Do1ADEF, Kristi A. Njaravelil1ABDF, Kwabena Ayesu1ADF, Mario Madruga1ABDE, Steve J. Carlan 23DEF*
Am J Case Rep 2023; 24:e941731
BACKGROUND: Ludwig angina is a cellulitis of the soft tissues of the neck and floor of the mouth. It is most commonly caused by Viridans streptococcal species, but other bacterial species have been shown to lead to this severe infection. Clostridium sporogenes is an anaerobic gram-positive, spore-producing bacillus found in soil and the human gastrointestinal tract. This report is of a case of a 49-year-old HIV-positive man with alcoholism and poor dental hygiene leading to a molar abscess who presented with Ludwig angina due to C. sporogenes.
CASE REPORT: A 49-year-old man presented with severe left molar pain, fever, and worsening neck swelling for 5 days. His medical history was significant for AIDS; he was not on antiretroviral therapy. Computed tomography of the neck was positive for extensive subcutaneous emphysema of the left sublingual space. Ludwig angina was diagnosed, and he was taken urgently for incision and drainage of the bilateral neck fascial space. On day 6 of hospitalization, 1 of 2 blood cultures grew C. sporogenes. He left the hospital on day 13 and was readmitted 6 days later with progression of the disease and alcohol withdrawal.
CONCLUSIONS: This case illustrates the need for rapid diagnosis and treatment of Ludwig angina and the importance of considering commonly pathogenic and rarely pathogenic bacteria when considering the underlying bacterial cause of an infection in an immunocompromised patient. To the best of our knowledge, this is the first case of Ludwig angina caused by C. sporogenes reported in the medical literature.
Keywords: Clostridium Infections, HIV Infections, Neck
Ludwig angina is a cellulitis of the submandibular, submental, and sublingual regions, which can progress quickly . Rapid identification of the disease and accurate treatment of its underlying cause, therefore, become vital components of proper management and reduced morbidity and mortality . Typical clinical presentation of Ludwig angina can include painful neck swelling, tooth pain, dysphagia, dyspnea, fever, tongue protrusion, tongue elevation, and malaise . Prior to initiating antibiotic treatment or surgery, the patient’s airway must be secured, as airway compromise is the leading cause of death for this disease . After securing the patient’s airway, the criterion standard of imaging for evaluation of disease severity is computed tomography (CT) . Typical CT findings of Ludwig angina of the neck can include muscle edema, focal fluid collections, gas in the soft tissue space, thickening of the soft tissues, and increased attenuation of the subcutaneous fat . Management consists of broad-spectrum antibiotic coverage, possible surgical drainage of the infection site for suppurative infection, and close airway monitoring [2,3].
Previously, the mortality rate of Ludwig angina was 50%, but modern advancements in antibiotics, imaging, and surgical techniques have reduced the mortality rate to 8% . However, comorbidities associated with the development of further complications include diabetes mellitus and HIV infection . One such complication is infection with rare pathogens, such as
A 49-year-old man presented to the Emergency Department with concerns of severe left molar pain, fever, and worsening neck swelling for 5 days. At the time of presentation, the patient confirmed foul-tasting drainage in his mouth, congestion, sore throat, cough, shortness of breath, neck pain, intermittent fevers, and chills. His past medical history was significant for AIDS, which was not treated with antiretroviral therapy, poly-substance abuse, and depression. He tested HIV-positive 6 years prior to presentation and had remained compliant with his prescribed combined emtricitabine, rilpivirine, and tenofovir alafenamide (Odefsey) until he ran out 2 months prior to presentation and was unable to afford refills. His polysubstance abuse consisted of smoking 0.25 packs of cigarettes daily, frequent cocaine and marijuana use, and weekly intake of 60 cans of beer and 3 shots of liquor. No other significant comorbidities were noted in his past medical history. His family history was unremarkable.
Physical examination was positive for muffled voice quality, poor dentition, minimal ability to open his mouth, severe pain on palpation of left posterior molar, and severe swelling, in-duration, and tightness of the anterior neck. There was crepitus on palpation of the area. On initial presentation, the patient was febrile, with a temperature of 38°C, had tachycardia, with a heart rate of 93 beats per min, had tachypnea, with a respiratory rate of 23 breaths per min, and had a blood pressure of 107/67 mm Hg. His oxygen saturation was 95% on room air, and he was not in acute respiratory distress. His initial laboratory test results were significant for leukocytosis, with a white blood cell count of 14.7×103/uL (reference range: 4.4–10.5×103/uL), anemia, with hemoglobin level of 11.7 g/dL (reference range: 11.4–14.7 g/dL), hyponatremia, with sodium level of 125 mmol/L (reference range: 136–145 mmol/L), and an acute kidney injury, with a blood urea nitrogen level of 41 mg/dL (reference range: 7–25 mg/dL) and creatinine level of 1.59 mg/dL (reference range: 0.60–1.20 mg/dL). His lactate level was within normal limits, and his viral respiratory panel was negative. His absolute clusters of differentiation 4 (CD4 T cell) count was 260.56 cells/uL (reference range: 492.00–1656.00 cells/uL), and his HIV viral load was 109 000. Due to concern for infection, blood cultures were drawn on initial presentation.
Computed tomography (CT) of the neck was positive for extensive subcutaneous emphysema of the left sublingual space, extending inferiorly to the bilateral submental regions and diffusely along the fascial planes of the anterior neck down to the level of the anterior superior mediastinum. There was no evidence of a discrete walled-off fluid collection. A dental cavity with associated periapical lucency involving the posterior-most left mandibular molar with evidence of cortical breakthrough of the lingual cortex was also seen and noted as a possible source of infection (Figure 1).
Based on the above findings, a diagnosis of Ludwig angina with extensive bilateral deep soft tissue edema of the neck with emphysema secondary to a dental abscess was made. He was taken urgently by the Oral Maxillofacial Surgery Department for incision and drainage of the bilateral neck fascial space, extraction of the third molar on the lower left side (tooth #17), and excision of any abnormal appearing or inflamed mandibular bone surrounding tooth #17. Perioperatively, incision of the left submandibular region and subsequent blunt dissection revealed left submandibular and sublingual abscesses, which were cultured, and sublingual fluid loculations along the subperiosteal plane of the lateral and medial cortices of the mandibular bone. Additional incisions and blunt dissection toward the inferior border of the mandible and the anterior neck revealed abundant purulent drainage in the bilateral sub-mental, left submandibular, left sublingual, and anterior neck spaces. Abnormal-appearing bone surrounding tooth #17 was excised. Three drains were placed in the left sublingual space, anterior neck, and submental space. He was then started on vancomycin, ceftriaxone, and clindamycin but his treatment was later de-escalated to ampicillin/sulbactam and vancomycin per the recommendations of the Infectious Disease team. He was also started on antiretroviral therapy, with 200 mg of emtricitabine, 25 mg of rilpivirine, and 25 mg of tenofovir alafenamide (Odefsey). His acute kidney injury and hyponatremia resolved during his stay.
The blood cultures drawn during the patient’s initial presentation took 43 h to identify the causal organism, with 1 culture initially showing anaerobic gram-positive bacilli. On day 6 of hospitalization, 1 of 2 blood cultures grew pan-susceptible
Six days later, the patient presented again to the Emergency Department due to continued wound drainage and skin burning in that area. He had not been compliant with his oral antibiotic treatment. CT of the neck revealed diffuse soft tissue edema and inflammatory changes, with an apparent open wound in the left anterior neck. There was also airway effacement, particularly in the region of the oropharynx. No focal walled-off collections were found (Figure 2).
He underwent elective endotracheal intubation due to severe airway compromise, was brought back to the operating room for surgical washout and complex repair of his anterior neck wound, and was found to have healthy granulation tissue in the bilateral neck wound bed with no purulent drainage found. He was then extubated and transferred to the Intensive Care Unit. Repeat blood cultures collected at the time of readmission had no growth. His course was further complicated by alcohol withdrawal seizures requiring emergent tracheostomy due to severe swelling in the posterior oropharynx, leading to the inability to visualize the vocal cords for re-intubation. He also developed severe dysphagia due to this severe oropharyngeal edema, requiring percutaneous endoscopic gastrostomy (PEG) placement. He was treated and cleared for discharge by the Pulmonology Department to a long-term care facility after a prolonged hospital stay.
Ludwig angina can be rapidly fatal if not quickly identified and treated, particularly if it is caused by a rare pathogen in an immunocompromised patient. The infection of Ludwig an-gina is typically polymicrobial and is most commonly caused by Viridans streptococci species. Other common causal organisms include staphylococcus species, peptostreptococcus, fusobacterium, bacteroides, and actinomyces .
Our patient did not grow any of the common causal organisms of Ludwig angina in his blood cultures. Instead, he grew
There are very few reported cases of severe infection caused by
Several previous case reports of infection caused by
Our patient’s course was complicated by his noncompliance with medication upon leaving against medical advice and later tracheostomy and PEG placement due to alcohol withdrawal seizures. It is well documented that alcohol withdrawal seizures are much more likely to lead to tracheostomy and PEG placement . Our patient’s noncompliance with his antibiotics led to a situation in which re-intubation was not possible due to severe edema of the oropharynx, thus necessitating emergent tracheostomy placement. In addition, this same oropharyngeal edema caused our patient to have severe dysphagia, leading to PEG placement. Our patient’s course therefore highlights the extreme importance of the role the patient plays in managing their own care. When faced with an immunocompromised patient whose compliance is in question, it is of the utmost importance for the physician to educate the patient on the possible risks of leaving against medical advice, including but not limited to the possibility of a prolonged and complicated disease course.
It is not clear how
There is no current criterion standard of treatment for
Most cases of Ludwig angina are caused by pathogenic bacteria, such as Viridans streptococcal species and occasionally staphylococcus and actinomyces. Less pathogenic bacteria have been reported in cases when there are comorbidities affecting the immune response, as in this case in which
FiguresFigure 1.. Computed tomography of the head and neck. There is extensive subcutaneous emphysema (red arrows) extending through the bilateral submental regions and along the fascial planes of the anterior neck to the level of the superior mediastinum, frontal, and sagittal views. Figure 2.. Follow-up computed tomography of the neck. There is diffuse soft tissue edema and inflammatory changes with an apparent open wound in the left anterior neck with some adjacent soft tissue gas pockets (red arrows) tracking to the left supraclavicular region that may be postsurgical and airway effacement, particularly in the region of the oropharynx.
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