Abstract
Introduction and importance of case
Ludwig's angina is an inflammation of neck spaces making it immediately life-threatening. The infection spreads to adjacent planes destructing facial planes, aspirations of infective particles, or septic embolism to distant areas. Understanding the rare presentations will help early diagnosis and treatment.
Presentation of case
This is about a 40 years-old man who presented with painful anterior neck swelling of 7 days duration. A diagnosis of Ludwig's angina with unilateral facial nerve paralysis and treated with immediate incision and drainage.
Clinical discussion
Ludwig's may present clinical with a variety of complications. This complication may be related to ongoing sepsis or mass effects manifesting with airway compromise or nerve palsy.
Conclusion
Although facial nerve palsy associated with Ludwig's angina is rare, it improves with immediate surgical decompression.
Keywords: Ludwig's angina, Facial nerve palsy, Airway obstruction, Case report
Highlights
-
•
The trend in incidence of Ludwig's angina is on decline.
-
•
Ludwig's angina is fatal unless intervened early.
-
•
Ludwig's angina has rare and atypical features with facial nerve compression.
-
•
Early operation is important for the improvement of facial nerve palsy.
1. Introduction
Ludwig's angina is an inflammation of neck spaces that easily spreads destructing adjacent planes making it immediately life-threatening [1], [2]. Here, we presented and incorporated management approaches on a case of Ludwig's angina with unilateral facial nerve paralysis with the involvement of its all branches. The case report adheres to SCARE criteria [3].
2. Case presentation
A 40-year-old man presented with painful anterior neck swelling and low-grade intermittent fever of 7 days duration. The swelling was progressive and developed difficult mouth opening & drooling of saliva on the left side after 4 days of initial symptom onset. However, he has no history of tooth decay or known chronic illness (Fig. 1).
Fig. 1.
Illustration showing swelling in the submandibular region bilaterally.
On examination, there was a 9 × 7 cm tender swelling of the sub-mandibular, sub-mental, and superior part of the anterior neck with palpable crepitus and erythematous overlying skin. He was unable to frown or close the eye and flat nasolabial fold on the left side. A clinical diagnosis of Ludwig's Angina was made.
The white blood cell count was 9600/mm3 with hemoglobin of 15 g/dl. There was no CT scan for better imaging. Thus, ultrasound scanning was done & showed diffusely edematous changes over the bilateral submandibular, sub-mental, floor of the mouth, and anterior cervical spaces.
He was started with crystalloids, ceftriaxone and metronidazole, and analgesics and operated to drain 100 cm3 foul-smelling thick pus. On day-6, he developed cough & generalized body swelling for which echocardiography was done & revealed 2.3 cm depth of pericardial effusion. Pericardioscentesis cytology showed reactive effusion while pleural fluid aspirate revealed gram-positive cocci on microscopy. In addition, serum Creatinine was high (4.2 g/dl). Diagnosis of acute kidney injury and hospital acquired pneumonia was added. Subsequently, antibiotics were revised to renal adjusted dose of vancomycin, ceftazidime and metronidazole to be completed over two weeks. The patient was discharged to home after 3 weeks with edema subsided and renal insult resolved. On subsequent follow up after 2 months, wound had healed and had improvement of facial asymmetry. At the 6th month, evaluation showed no more facial palsy.
3. Clinical discussion
Ludwig's angina is an inflammation of multiple neck spaces which makes it immediately life-threatening [1]. It spreads to planes adjacent to the airway making a timely decision important [2].
Although the trend of incidence is on the decline [4], some segments of a population with compromised immunity may acquire a serious infection [5]. Etiologic agents are inclusive of Streptococcus sp., Staphylococcal aureus, Haemophilus influenza, and some anaerobes.
Depending on the etiologic type and/or immune status of the individual, clinical progress may be different. It may spread very rapidly to compromise the airway [6], [7]. The management of patients with Ludwig's angina is urgent surgical decompression to halt further spread and complications [8].
The disease may have a rarer complicated course with neck necrotizing fasciitis [9], extra pleural empyema thoracic [10], or mediastinitis [11]. Kissig et al. described clinical findings from 35 cases of paralysis of facial nerve related to iatrogenic, tumor, trauma, congenital, idiopathic, and inflammation causes [12]. However, none was with all branches of facial nerve involvement. Apart from septic focus spread, Ludwig's angina may result in facial nerve palsy due to compression of the nerve trunk in narrow compartment in its course through muscles and confined tissue spaces manifested by recurrent laryngeal nerve paralysis [13], and marginal mandibular nerve palsy [14]. In this particular case, the complication could have arisen due to the involvement of the facial nerve secondary to the inflammatory process or compression at its exit from narrow stylomastoid foramen. This pathogenesis is the basis for urgent surgical decompression of swelling with or without steroid administration [15].
4. Conclusion
Although, clear delineation of the anatomy may be challenging in the absence of advanced imaging (CT scan or MRI), early clinical diagnosis and surgical decompression of Ludwig's angina is important to control ongoing sepsis and improve clinical outcome.
Consent
Before data collection, written informed consent was acquired and the laboratory procedure was done with the essence of beneficence and data were kept confidential. The written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of written consent is available for review by the Editor-in-Chief of this journal on request.
Ethical approval
The study is approved by the Research Review Committee of the institution (referenced: S/R/C/36/01/23). Before data collection, written informed consent was acquired and the laboratory procedure was done with the essence of beneficence and data were kept confidential.
Funding
There is no funding received for this work.
Author contribution
Megbar Dessalegn: Writing – original draft, review & editing.
Manendante Bogale: Writing – original draft, review & editing.
Dawit Alemayehu: Review & editing.
Wubshet Assefa: Review & editing.
Tilahun Deresse: Review & editing
Guarantor
Megbar Dessalegn.
Research registration number
Not applicable.
Declaration of competing interest
There is no conflict of interest.
References
- 1.Doldo G., Albanese I., Macheda S., Caminiti G. Ludwig angina: a disease of the past century. Case report. Minerva Anestesiol. 2001;67(11):811–814. [PubMed] [Google Scholar]
- 2.Grodinsky M. Retropharyngeal and lateral pharyngeal abscesses: an anatomic and clinical study. Ann. Surg. 1939;110(2):177. doi: 10.1097/00000658-193908000-00003. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 3.Agha R.A., Franchi T., Sohrabi C., Mathew G., Kerwan A. The SCARE 2020 guideline: updating consensus surgical CAse REport (SCARE) guidelines. Int. J. Surg. 2020;84:226–230. doi: 10.1016/j.ijsu.2020.10.034. [DOI] [PubMed] [Google Scholar]
- 4.Fritsch D., Klein D. Ludwig's angina. Heart Lung. 1992;21(1):39–46. [PubMed] [Google Scholar]
- 5.Flint P.W., Haughey B.H., Robbins K.T., Thomas J.R., Niparko J.K., Lund V.J. Elsevier Health Sciences; 2014. Cummings Otolaryngology-head and Neck Surgery e-book. [Google Scholar]
- 6.Mendelson M.H., Gurtman A., Szabo S., Neibart E., Meyers B.R., Policar M., et al. Pseudomonas aeruginosa bacteremia in patients with AIDS. Clin. Infect. Dis. 1994;18(6):886–895. doi: 10.1093/clinids/18.6.886. [DOI] [PubMed] [Google Scholar]
- 7.Infante Cossío P., Fernández Hinojosa E., Mangas Cruz M.Á., González Pérez L.M. 2010. Ludwig’s Angina and Ketoacidosis as a First Manifestation of Diabetes Mellitus. [DOI] [PubMed] [Google Scholar]
- 8.Movva H., Taksande K. Anaesthetic challenges in a patient of Ludwig's angina: a case report. Cureus. 2022;14(10) doi: 10.7759/cureus.30570. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 9.Chueng K., Clinkard D.J., Enepekides D., Peerbaye Y., Lin V.Y.W. An unusual presentation of Ludwig’s angina complicated by cervical necrotizing fasciitis: a case report and review of the literature. Case Rep. Otolaryngol. 2012;2012 doi: 10.1155/2012/931350. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 10.Young J.N., Samson P.C. Extrapleural empyema thoracis as a direct extension of Ludwig's angina. Case report. J. Thorac. Cardiovasc. Surg. 1980;80(1):25–27. [PubMed] [Google Scholar]
- 11.Furst I.M., Ersil P., Caminiti M. A rare complication of tooth abscess-Ludwig's angina and mediastinitis. J. Can. Dent. Assoc. 2001;67(6):324–329. [PubMed] [Google Scholar]
- 12.Kissig B., Zierz S. Isolated pareses of the terminal branches of the facial nerve: clinical findings, etiology and differential diagnosis. Fortschr. Neurol. Psychiatr. 2006;74(7):371–376. doi: 10.1055/s-2005-915579. [DOI] [PubMed] [Google Scholar]
- 13.Pérez D., SH J.A. Bilateral recurrent nerve paralysis secondary to Ludwig's angina. Acta Otorrinolaringol. Esp. 1999;50(2):172–174. [PubMed] [Google Scholar]
- 14.Sarkar D., Sarkar S., Mangalgiri A.S., Thakur G., Beohar V. 2010. Marginal Mandibular Nerve Palsy as an Unusual Complication in Ludwig’s Angina. [Google Scholar]
- 15.Kim S.J., Lee H.Y. Acute peripheral facial palsy: recent guidelines and a systematic review of the literature. J. Korean Med. Sci. 2020;35(30) doi: 10.3346/jkms.2020.35.e245. [DOI] [PMC free article] [PubMed] [Google Scholar]