Mediastinitis, Pericarditis, Empyema, and Bacteremia: An Infrequent Complicated Ludwig's Angina Case

Diyaa Hassan Bokhary; Abdulmoneim Ali Mustafa; Ghada Asad Merdad

doi:10.4103/sjoh.sjoh_41_22

CASE REPORT

Year : 2022 | Volume : 24 | Issue : 4 | Page : 182-184

Mediastinitis, Pericarditis, Empyema, and Bacteremia: An Infrequent Complicated Ludwig's Angina Case

Diyaa Hassan Bokhary, Abdulmoneim Ali Mustafa, Ghada Asad Merdad
Department of Emergency Medicine, King Abdul-Aziz University Hospital, Jeddah, Saudi Arabia

Date of Submission	04-Sep-2022
Date of Decision	12-Oct-2022
Date of Acceptance	13-Oct-2022
Date of Web Publication	30-Dec-2022

Correspondence Address:
Dr. Diyaa Hassan Bokhary
King Abdul-Aziz University Hospital, Jeddah
Saudi Arabia

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/sjoh.sjoh_41_22

Abstract

Ludwig's angina is a life-threatening cellulitis of the submandibular, submental, and sublingual areas that originates mostly from odontogenic illnesses. It is associated with high morbidity and mortality rates. Its natural course, if not treated, leads to the rapid progression and development of complications, including airway compromise, necrotizing fasciitis, septic shock, pericarditis, and empyema. We report the case of a 40-year-old male who presented primarily with chest pain associated with cheek pain, sore throat, odynophagia, and subjective fever. The patient was diagnosed with Ludwig's angina secondary to a poor oral hygiene complicated by pericarditis, mediastinitis, and left-sided empyema. An immediate antibiotic administered, and subsequently neck dissection with left anterolateral thoracotomy was performed. In this case report, we highlight the importance of not only early recognition but also rapid intervention to prevent disease progression, including early empiric antibiotics, airway management, and surgical intervention, especially if the patient presents with an atypical or vague complaint.

Keywords: Ludwig's angina, mediastinitis, odontogenic illnesses, pericarditis

How to cite this article:
Bokhary DH, Mustafa AA, Merdad GA. Mediastinitis, Pericarditis, Empyema, and Bacteremia: An Infrequent Complicated Ludwig's Angina Case. Saudi J Otorhinolaryngol Head Neck Surg 2022;24:182-4

How to cite this URL:
Bokhary DH, Mustafa AA, Merdad GA. Mediastinitis, Pericarditis, Empyema, and Bacteremia: An Infrequent Complicated Ludwig's Angina Case. Saudi J Otorhinolaryngol Head Neck Surg [serial online] 2022 [cited 2023 Jan 30];24:182-4. Available from: https://www.sjohns.org/text.asp?2022/24/4/182/366333

Introduction:

Ludwig's angina is an extensive infection and inflammation of the submandibular, submental, and sublingual anatomical spaces.^[1] It is considered one of the most emergent otolaryngologic (ENT) diseases, as it carries a high mortality rate ranging from 17.7% to 41%, even if recognized and treated promptly.^[2] In addition, it can rapidly progress and spread to the neck, mediastinum, and rarely to thoracic visceral structures. This condition most commonly originates from dental illness; however, other nondental conditions have been reported to be risk factors for Ludwig's angina, including poor nutrition, injectable drug abuse, diabetes, systemic lupus erythematosus, and immunocompromise.^[1]

In 1980, a case of Ludwig's angina complicated by empyema, pulmonary infiltration, and pericarditis was reported by Strauss.^[3] In addition, some other scattered case reports were found in the literature, but most lack good quality illustrative images. Here, we report a case of Ludwig's angina, complicated with neck and mediastinal extension, pericarditis, and left-sided chest empyema, in a male patient with no medical history of note.

Case Report

A 40-year-old Somali male patient with no known medical or surgical illnesses, working as a truck driver, presented to our emergency department (ED) with a history of chest pain radiating to the left shoulder, which worsened with deep respiration and coughing. A more comprehensive history revealed that he had experienced a subjective fever and progressive sore throat for 3 days causing decreased oral intake with no hoarseness or difficulty breathing. He also had a history of tooth pain a few months prior, but no previous dental procedures.

On assessment, his vital signs were heart rate of 111 beats/min, blood pressure of 150\95 mmHg, respiratory rate of 20 breaths/min, oxygen saturation of 99% on room air with no signs of respiratory distress, and a temperature of 37°C. On assessment, he was irritable, in severe pain, and had a poor oral hygiene. The left cheek, submandibular area, and neck were swollen, tense, red, hot, and tender.

Chest examination revealed bilateral equal air entry with no added sounds. There were no cardiac murmurs or friction rubs on cardiac auscultation. Electrocardiogram showed sinus tachycardia and diffuse ST-segment elevation in most leads, with ST-segment depression and PR elevation in the aVR lead [Figure 1]. After our ED assessment, we suspected Ludwig's angina as the most likely differential diagnosis. Therefore, the patient was immediately considered for nothing per mouth (NPO), started empirically on piperacillin-tazobactam and administered dexamethasone.

Figure 1: Electrocardiography showing sinus tachycardia and diffuse ST-segment elevation in most leads, with ST-segment depression (red arrow) and PR elevation (black arrow) in the aVR lead. PR: PR-segment

Click here to view

Laboratory evaluation showed a white blood cell count of 13.54/mm³ (reference range: 4.5–11.5/mm³) with a neutrophil predominance of 87.9% (reference range: 50%–70%) and lactate level of 2.2 mmol/L (reference range: 0.4–2 mmol/L). Troponin-I was 1.4 ng/ml (reference range: <0.4 ng/ml), and there was the anterior rim of a small pericardial collection on bedside cardiac echocardiography, which suggested pericarditis with effusion.

Initial chest X-ray revealed cardiomegaly. A fiberoptic evaluation was performed to assess the airway, which showed inflamed edematous hypopharynx and larynges with obvious mobile vocal cords and visible arytenoids. Computed tomography (CT) was performed and demonstrated superficial and deep neck emphysema and abscess, dissecting down to anterior mediastinum, with pneumomediastinum, pneumopericardium, and a small pericardial effusion [Figure 2]a and [Figure 2]b.

Figure 2: (a) Cross-sectional, computed tomography scan of the chest with intravenous contrast, showing pneumopericardium (white arrow) and pericardial effusion (black arrow). (b) Sagittal view, computed tomography scan of the neck with intravenous contrast, showing superficial and deep neck emphysema (white arrow) and abscess, dissecting down to mediastinum, with pneumomediastinum

Click here to view

At this point, the diagnosis of Ludwig's angina was confirmed, and ENT and anesthesia consultation were requested immediately. In addition, cardiology consultation was performed for the associated pericarditis. Critical airway equipment were requested, prepared and have been set on the bedside patient's table. After which the patient was quickly shifted from ED directly to the Operation Room (OR), with an ENT and anesthesia physicians, where a neck exploration procedure was performed, which showed pus collection and widespread inflammation extending down to the chest. Three Penrose drains were inserted and samples were sent for culture. The patient was then transferred to the surgical intensive care unit (SICU).

The next day, the patient was retaken to the OR by the thoracic surgery team, where he was found to have an anterior mediastinal pus collection and pericardial effusion. He underwent left anterolateral thoracotomy, mediastinal drainage, left pleural decortication, and pericardial window. Chest radiography was repeated after the second procedure and showed left pleural effusion [Figure 3], which was drained by a thoracostomy tube, and samples were sent for the analysis and culture.

Figure 3: Chest X-ray showing bilateral pleural effusion more severe in the left side (white arrow)

Click here to view

Cultures from the neck and mediastinal collection grew Pseudomonas aeruginosa and Streptococcus anginosus. Anaerobic peripheral blood culture grew Streptococcus constellatus. Moreover, the pleural fluid culture also grew S. constellatus. Therefore, an infectious disease (ID) team was consulted to review all culture results and bacterial antimicrobial sensitivity, and the patient was administered ceftazidime, azithromycin, and metronidazole. In addition, proper daily wound dressing changes and wound care were strictly applied and continuously evaluated.

A few days later, CT chest with contrast showed an interval decrease in the mediastinal and pericardial fluid collection. The patient's overall condition improved gradually over 1 month. The surgical drains and chest tube were removed; the neck wound approximated except for 1 cm left unclosed to allow packing, and then, the patient was discharged on a 4-week antibiotic course of ciprofloxacin and clindamycin, and outpatient department clinic appointments with ENT, thoracic surgery, and ID after 4 weeks, and daily dressing clinic appointments for wound assessment and care.

Discussion

In 1836, Wilhelm Frederick von Ludwig described the sublingual area's first case of extensive cellulitis.^[1] The most common risk factors are dental infection or recent dental procedure in approximately 90% of the cases.^[4] However, even in immunocompetent patients, a poor oral hygiene alone is considered a causative etiology for Ludwig's angina, as in our case.^[4] The patient in this case complained of the left lower molar tooth pain for about 6 months, and extensive dental caries were found on clinical examination. However, he did not seek dental care, and no dental manipulation was performed.

It is typically known to be an acute, rapidly progressive disease, in which the patient most commonly presents with submandibular and neck pain, sore throat, dysphagia, and trismus.^[3] The commonly reported complications include airway compromise, necrotizing fasciitis, and septic shock.^[5] In concordance with what was found in our patient, a case report of a patient from 1980 described complicated Ludwig's angina presenting with pericarditis and empyema.^[3] Most recently, in May 2005, an elderly patient presented to the ED with a complaint of chest pain and respiratory distress; a few hours later, he started to develop neck swelling and was discovered to have Ludwig's angina, after he underwent neck and chest CT scan.^[6] In addition, mandibular osteomyelitis, another rare complication of Ludwig's angina, has been reported in the literature.^[7]

The most isolated organisms are Streptococcus, Staphylococcus, and anaerobic organisms such as Bacteroides, Peptococcus, Fusobacterium, and Peptostreptococcus.^[8] Other rare organisms have been reported in the literature, including Corynebacterium diphtheriae, Pneumococcus, and Salmonella ^{More Details} Typhi.^[9] In addition, Pseudomonas, Candida, Escherichia ^{More Details} coli, and Clostridium are atypical organisms found mainly in immunocompromised patients. In our patient, P. aeruginosa and S. anginosus were isolated from the drained neck and mediastinal collections; however, our patient was not immunocompromised.

The natural course of Ludwig's angina involves the diffuse extension and rapid progression. Therefore, an early antibiotics administration such as penicillin G, metronidazole, and clindamycin should be started empirically to cover Gram-positive, Gram-negative, and anaerobic bacteria. Furthermore, an immediate source control by incision and drainage is essential to limit infection progression and avoid possible complications.^[10]

In conclusion, Ludwig's angina is a life-threatening disease with high morbidity and mortality rates. As health-care practitioners, we infrequently face the cases of Ludwig's angina due to improvements in the general awareness of the importance of maintaining good oral hygiene and increasing antibiotic availability. By reporting such a case, we highlight the importance of not only early recognition but also rapid intervention to prevent disease progression, including early empiric antibiotics, airway management, and surgical intervention, especially if the patient presents with an atypical or vague complaint.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initial s will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	Pak S, Cha D, Meyer C, Dee C, Fershko A. Ludwig's angina. Cureus 2017;9:e1588.
2.	Dajer-Fadel WL, Borrego-Borrego R, Flores-Calderón O, Argüero-Sánchez R, Navarro-Reynoso FP, Ibarra-Pérez C. Descending necrotizing mediastinitis associated with spinal cord abscess. Asian Cardiovasc Thorac Ann 2013;21:90-2.
3.	Strauss HR, Tilghman DM, Hankins J. Ludwig angina, empyema, pulmonary infiltration, and pericarditis secondary to extraction of a tooth. J Oral Surg 1980;38:223-9.
4.	Shemesh A, Yitzhak A, Ben Itzhak J, Azizi H, Solomonov M. Ludwig angina after first aid treatment: Possible etiologies and prevention-case report. J Endod 2019;45:79-82.
5.	Balasubramanian S, Elavenil P, Shanmugasundaram S, Himarani J, Raja VK. Ludwig's angina: A case report and review of management. SRM Journal of Research in Dental Sciences 2014;5:211.
6.	Ocasio-Tascón ME, Martínez M, Cedeño A, Torres-Palacios A, Alicea E, Rodríguez-Cintrón W. Ludwig's angina: An uncommon cause of chest pain. South Med J 2005;98:561-3.
7.	Otsuka Y, Harada K, Nishimura Y, Otsuka F. Ludwig's angina and mandibular osteomyelitis after dental extraction. JMA J 2019;2:87-8.
8.	Parker E, Mortimore G. Ludwig's angina: A multidisciplinary concern. Br J Nurs 2019;28:547-51.
9.	Mahajan RK, Sharma S, Madan P, Sharma N. Ludwig's angina by salmonella typhi: A clinical dilemma. Indian J Med Microbiol 2015;33:300-2. [PUBMED] [Full text]
10.	Kovalev V. A severe case of Ludwig's angina with a complicated clinical course. Cureus 2020;12:e7695.