We report a case of Ludwig Angina with DNM observed at the end of December 2019. A healthy 17-year-old female was admitted to the paediatric intensive care unit at Cartage Clinic, Tripoli, Libya, with a history of swelling in neck, tongue and lips for two weeks (Fig.1).
On the initial examination, the patient was leaning forward, had shortness of breath and complaining of dysphagia, aphonia, and regurgitation even with water through the nose. The patient couldn't lie on the bed, slept on upright position with orthopnea. She was febrile over 38 ⁰C and recorded a heart rate of 95-110 b/m, blood pressure of 124/70 mm Hg, respiratory rate of 35-38 b/m, and an oxygen saturation of 89-92%. Examination revealed the anterior fullness of the neck, with a deep purplish discoloration of her skin.
There was significant swelling, and induration in the submandibular and submental regions extending down towards the base of the neck, with marked decrease in air entry on the right side of the lung, other physical examination was unremarkable.
The ethics statement, After the family is informed of the treatment options (surgical and medical), they accepted tooth extraction and aspiration of the collected fluid in submandibular and plural areas besides the medical treatment.
The study was approved by the Ethics Committee (Bioethics committee at Biotechnology Research Center N⁰: BTC-BTRC 24_2021). An informed consent was obtained for the family patient; and the study was carried out according to Helsinki declaration.
Laboratory test and imaging
The diagnosis was DNM arising from Ludwig’s Angina with a dental infection source. Clinical features and presentations, supported by the laboratory and radiology exams are requested for diagnostic assistance such as; complete blood count (CBC), renal function test, tuberculin skin test (TST), Immunoassay and tumor markers (Table 1).
We did an ultrasound, computed tomography (CT) scans of the neck, chest x-ray, chest, and abdomen. We collected samples from both submandibular and plural areas for biochemistry, culture, and sensitivity (C/S) analysis (Table 2,3).
The laboratory report of the fine needle aspiration samples submandibular swelling shows that cytological features are consistent with acute purulent inflammation. In the details we can say that the samples show mixed inflammatory cells composed of many neutrophils, small lymphocytes, and many foamy macrophages, in the background of fibrin, cellular debris, and red blood cells. Multiple scattered mature squamous epithelial cells are seen. No multinucleated giant cells are seen. No atypical or malignant cells are seen in both samples of pleural and submandibular swelling (Table 2,3).
Furthermore, laboratory data was significant for a leukocyte count of 19.1 n x 103 cells/µl, C-reactive protein of 96 mg/dL. And noted that the HIV screen was negative.
Chest x-ray showing massive right pleural effusion and blunting of the costophrenic angle (Fig.2). An ultrasound scan of the submandibular area and neck swelling showed right parotid gland hypertrophy with intraparotid massive abscess with submandibular turbid collection and multiple inflammatory lymphadenopathy.
The first CT scan of the neck and chest was 1st week of admission show a large well-defined lobulated collection involved bilateral parapharyngeal spaces, prevertebral cervical space, tracking down beneath platysma muscle bilateral submandibular region of the anterior and posterior mediastinum associated with right-side pleural effusion.
On 2nd CT SCAN of the neck and chest with contrast which was at the 4th week of admission (Fig. 3 A) showing in comparison with previous CT scan of the 1st week of admission. The comparison shows that there is a newly developed small collection socket on the right side of the nasopharynx 2.0 cm and 1.6 cm posteriorly to the upper trachea. The unchanged collection is just below the level of the thyroid gland in the anterior lower neck soft tissue.
A newly developed small socket collection in the posterior mediastinum 3.3 cm in the axial diameter and unchanged collection in the anterior mediastinum 7.3 x 2.7 cm with some new post interventional air inside.
Post removal of the right-side chest tube with newly developed mild right-side pleural effusion, no evidence of suspected Pneumothorax, Nasopharynx, oropharynx, and larynx appear normal. However, Normal appearance of the bilateral thyroid gland lobes. Normal scanned the rest of the bilateral lung parenchyma. Trachea and main bronchi are normal in caliber.
The 3rd CT SCAN at 6th week of admission (Fig. 3 B) in comparison with previous CT scan in 4th week of admission shows dramatic regression of the previously seen multiple peripherally rim enhancing collections seen at upper and lower neck soft, and no more neck softly tissue collection can be detected.
Further, regression of the previously seen collection in the anterior mediastinum and right-side pleural effusion, there is just a minimal collection seen in the anterior mediastinum and small encysted pleural effusions are seen on the right side.
Once DNM was suspected, an experimental broad-spectrum intravenous antibiotic, including meropenem, vancomycin, metronidazole, and ampicillin as the first line of treatment was started for two weeks.
Our view is that administration of empirical antibiotics should cover aerobes and anaerobes for possible mixed infection. We then performed another CT scan as an assessment procedure, as CT of the Cervical thorax is the ideal image to evaluate the area of DNM involved. Especially, the aspiration of the collection in the submandibular region and right pleural effusion were performed under the complete aseptic technique at the 1st week of admission. The collection from the submandibular area with 500 ml of pus aspirated, sent to a laboratory for c/s (Fig.5).
The Pleurocentesis Chest tube guided by ultrasound was inserted and draining of 1250 ml of pleural fluid and sent to a laboratory for Analysis and c/s. Unfortunately, the second CT scan which was performed at the 4th week (after the aspiration) showed a new collection in the posterior mediastinum and newly developed mild right-side pleural effusion. We changed the antibiotic to Imipenem/ Cilastatin, Ceftriaxone, clindamycin, and Piperacillin/tazobactam this decision was made not according to any culture because it was aseptic collections (Table 2-3). However, extraction of the 3rd right molar tooth which has the dental caries was done, which was the source of infection, on the 10th day of admission.
The CT scan of the chest and neck was repeated in the 6th week, showing dramatic regression of the previously seen multiple peripherally rim enhancing collection at upper and lower neck. At the same time, no more neck soft tissue collection can be detected compared to the previous CT scan. The collection was completely resolved, and the patient completed 3 weeks of antibiotics after one month of discharge.