International Endodontic Journa

22, 142-147

Ludwig's angina: an unusual sequel to endodontic therapy

J, R, T\J\N* Department of Oral and Maxillofacial Surgery, Plymouth Greenhank Hospital, Plymouth, Devon
Summary, I,ud wig's angina is an unusual sequel to endodontic treatment: such a case is described. Principles of management are discussed with emphasis placed on the need to secure the airway early. The literature is reviewed.

Introduction Ludwig's angina is a rapidly progressive cellulitis involving bilateral sublingual, submandibular and submental spaces ofthe neck. Extensive brawny swelling develops with elevation and posterior displacement ofthe tongue and consequent airway obstruction. Preexisting acute dental infections in the form of abscesses, postextraction sepsis and pericoronitis are the most frequent causes, although infected fractures of the mandible, penetrating injuries of the neck and mouth, and suppurative sialadenitis have been described. It is believed that this is the first case reported to have followed endodontic therapy. Case report . A previously fit 5()-year-old timber merchant w as referred to the Oral Surgery service by the admitting house physician. The patient had a subacute airway obstruction and was unable to speak. He pointed to his tongue and to the left side of his mouth as the source of discomfort. The referring letter from his general practitioner stated that the patient had some 'conservative dental work' the preceding day and his tongue had subsequently become swollen. On the day of admission he had a rigor and now was unable to swallow water. The patient was sitting upright in bed and was pale, sweaty and pyrexial. He was normotensive and had a tachycardia. His tongue was Present address and correspondence: J, R, Tuffin, Department of Maxillofacial and Oral Surgery, North Manchester General Hospital, Crumpsall, Manchester, M8 6RB, UK,
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grossly swollen, being extruded from his mouth, forced between his teeth and impacted against his palate. He had no oral airway and could barely breathe through his nose. There was massive brawny swelling affecting bilateral sublingual and submandibular spaces producing displacement of his larynx and trachea to the right. The patient indicated that the swelling was increasing and hence a theatre was prepared for tracheostomy. Blood was taken for culture and antibiotic therapy started with cefuroxime and metronidazole. One hundred milligrams of hydrocortisone was given. The initial plan was to perform a tracheostomy under local analgesia but the opinion of an experienced senior anaesthetist was that intubation was possible with care. The patient was placed on the operating table and the surgeon scrubbed before induction of anaesthesia. After sedation with midazolam (5 mg) and topical anaesthesia of the right nasal passage, a 7,5-mm red rubber nasotracheal tube was passed into the nasopharynx. Anaesthesia was introduced using nitrous oxide and oxygen in a 50 per cent mixture and halothane. The tube was then passed into the trachea without difficulty thus relieving the obstruction. Marked hyperventilation and a brief period of atrial fibrillation followed presumably resulting from halothane uptake in the presence of hypercapnoea, Tracheostomy was performed without complication. Examination of his mouth was then possible. His tongue, floor of mouth and neck were grossly swollen (Fig, 1), The epithelium ofthe ventral surface of his tongue was bruised and stripping off (Fig, 2), Two bullae, 10 x 5 mm, were present on the right lateral border. The second left mandibular molar had a temporary restoration but was neither extruded nor mobile and no sign of ^djacent local inflammation was present. The remaining teeth were well conserved. His oral hygiene showed signs of

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Fig. 1. Condition after tracheostomy but prior to drainage: (a) face and (b) lateral view showing extensive submandibular and sublingual oedema.

Fig. 2. Grossly oedematous tongue showing stripping ofthe ventral epithelium.

recent neglect but his periodontium was adequately healthy. The healing socket of his left mandibular third molar was noted. Bilateral submandibular incisions delivered a small amount of serosanguinous fluid. Corrugated rubber drains were placed. The

second left mandibular molar was extracted and a small apical granuloma curetted from the socketthe goal was to eradicate any possible source of infection. In the endontically treated tooth, filling material was not extruded through the apex of the root nor were there

swelling had settled. He eventually made a full recovery. Investigations Blood cultures taken on admission grew Lancefield group A Streptococci sensitive to penicillin, amoxycillin and cefuroxime. Those repeated later showed no growth. Only respiratory organisms were cultured from the extracted tooth and from swabs taken from the tongue and drain sites. The white cell count on the first postoperative day was 12.1 x 10^/1, showing a left shift and toxic granulations. Fasting blood sugar was normal (5.1m mol/1). Radiographs of his jaws and teeth yielded no other cause for infection. Review of history Further details emerged as communication became possible. The patient had undergone little dental treatment during the preceding 6 years. Six weeks previously his left mandibular third molar had been removed and uneventful healing occurred. On the day prior to being admitted to hospital he had been well and had no sore throat. He attended his dental practitioner for treatment to his left mandibular second molar. The latter stated that analgesia had been achieved by means of two left inferior alveolar nerve blocks and no infiltrations were made into the tongue, buccal sulcus or gingiva. A 'partially vital dental pulp' had been extirpated. His dental surgeon had difficulty reaching the full length of the root canals but felt confident that no instrument had passed through any root apex. He took radiographs to establish the working lengths for instrumentation, after which he placed an endodontic dressing of demeclocycline and triamcinolone (Ledermix). Following this treatment the patient ate a sandwich whilst his mouth was still numb. He ate it carefully and thought that he had not bitten his tongue. The following morning he was aware of discomfort under the left side of his tongue. By midday he was pyrexial and had a rigor. His general practitioner felt the primary problem was septicaemia and hence referred him to the physicians. During the ambulance journey of approximately 1 hour his tongue swelled massively much to the surprise and concern of the ambulance crew.

Fig. 3. (Condition on the third postoperative day showing bilateral submandibular drains and occluded tracheostomv tube.

signs of local trauma. Swabs were taken from both drain sites, the tongue bullae and together with the extracted tooth were sent for bacterial culture.

Postoperative course Initially he was nursed sitting upright. His tongue was kept moist with saline and a rubber prop was retained in his mouth to prevent his tongue being traumatized by his teeth. Intravenous fluids and antibiotics were continued. By the second day, his tongue had reduced in size sufficiently for him to close his mouth, l'igure } shows his condition on the third postoperative day. On the fourth postoperative night his tracheostomy was occluded overnight without airway embarrassment and it was closed the following day under local analgesia. He left hospital 1 week after admission. On out-patient review he commented that he had a partial reduction in taste on the left side of his tongue but earlier symptoms of soreness resulting from the extensive oral

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Discussion In 1836 Wilhelm Freiderich von Ludwig described five cases with extensive cellulitis of the floor of the mouth and neck; in these patients gangrenous induration of the connective tissues of the neck advanced to involve the tissues which covered the small muscles between the larynx and the floor of the mouth (Patterson et al. 1982), Three out of five of these patients died, A hundred years later similar mortality figures were occurring (Williams 1940) but within a few years considerable improvement had been achieved. As a result of greater attention to airway preservation and the use of recently introduced sulphonamides, mortality figures fell to 10 per cent (Williams & Guralnick 1943), In 1940 Williams described eight cases which occurred in 1 year. However, Ludwig's angina is now uncommon; better dental care, improved antibiotics and further understanding of dentofacial infections have reduced its incidence.
Anatomical considerations

Infection may spread to the other tissue spacees of the neck and inferiorly to the mediastinum producing empyema, pulmonary infiltration and pericarditis (Strauss et al. 1980, Snow et al. 1983), Bounds (1985) reported extension of pus through the oesophageal hiatus resulting in a subphrenic abscess.
Bacteriology

Ludwig's angina may commence with infection in either the sublingual, submental or submandibular spaces. The infection opens the tissue spaces and allows pus to track along the tissue planes. Infection leading to Ludwig's angina usually starts in the submandibular spacethe most common cause being mandibular molar teeth and alveolus. The former have their roots close to the lingual plate of bone and their apices lie below the mylohyoid attachment thus affording access to the submandibular space (Tschiassny 1943). The submandibular space lies between the investing layer of the deep cervical fascia and the mylohyoid muscle. It contains the submandibular gland, lymph nodes and the facial artery and vein. It communicates with the submental space via the anterior belly of the digastric muscle and with the sublingual space around the posterior free border of the mylohyoid muscle. The sublingual spaces communicate with each other across the hump of the genial muscles. In the presence of severe inflammation with resulting oedema and hyperaemia the mylohyoid muscle presents a poor barrier to the spread of pus, thus enabling rapid communication between submandibular and sublingual spaces (Killey et al. 1975),

Before the development of antibiotics the most frequently isolated organisms in Ludwig's angina were Streptococci. Hought et al. (1980) reviewed 42 cases: the most common pathogenic organisms found were streptococci (42%), staphylococci (13%), bacteroides (6.2%) and Pseudomonas (6,2%). Gross et al. (1976) noted that mixed infections have become more common and drew attention to the importance of anaerobic bacteroides as the pathogen. In this case, Lancefield group A haemolytic streptococci were isolated from the initial blood cultures only. With P-haemolytic streptococcal septicaemia our patient was at risk from infective endocarditis which fortunately did not occur. Treatment The hallmark of Ludwig's angina is extensive brawny cellulitis rather than collections of copious pus. The intense oedema forces the tongue upwards and backwards and produces trismus and laryngeal oedema. Airway obstruction is of prime consideration. If the airway is threatened early tracheostomy, rather than as a last resort, is recommended, Tracheostomy under local analgesia is safer although this may be difficult since the inflammation produces pain on extension of the neck. Initially the swelling is superior to the hyoid and hence tracheostomy is not difficult, however, later the extensive oedema produces a difficult dissection with the trachea lying further from the skin. Dissection through grossly infected neck tissues may cause dissemination of organisms and inhalation pneumonia. General anaesthetic agents may cause further laryngeal irritation. Intubation is difficult and can be hazardous. Trismus, laryngeal oedema and displacement combined with the possibility of rupture of a thin walled abscess

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J. R. Tuffin susceptible than other cephalosporins to inactivation by penicillinases (British National Formulary 1988), Metronidazole is valuable against anaerobic organisms. In this case a combination of cefuroxime and metronidazole proved highly active, blood cultures being negative when repeated on the second postoperative day. This is a suitable front-line therapy for life-threatening infections which may be modified later when the results of a Gram stain and microbiological culture are available. Conclusion The exact cause of Ludwig's angina in this reported case is uncertain. The possible modes of infection are: (i) the dissemination of organisms from necrotic dental pulp into periapical tissues during endodontic therapy; (ii) the inoculation of streptococci existing in his mouth and pharynx prior to dental treatment into his tongue and sublingual tissues by the manipulation of instruments; (iii) an infected haematoma secondary to tongue biting; and (iv) unrelated. It is essential for all who provide dental care to realize that severe life-threatening dental sepsis may follow endodontic treatment. It is imperative that the principles underlying emergency management are understood. Acknowledgements I am indebted to Mr A, S, Davies, Consultant Oral Surgeon, for permission to report his case, and to Drs A, S, Davies and Julian Bion for their constructive criticism in the preparation of this paper. Also, I would like to thank the Department of Medical Photography, Derriford Hospital, Plymouth, References
BOUNDS, G,A,

with subsequent inhalation of infected exudate produce great risks, F'ailed intubation on an anaesthetized patient will necessitate emergency tracheostomya considerably more difficult procedure. Intubation, if chosen, should be undertaken with the patient placed on the operating table and the surgeon scrubbed ready to undertake emergency tracheostomy if necessary, Schwartz el al. (1974) described the use ofa Bbre-optic laryngoscope (although an intubating bronchoscope is now in more frequent use) over which the endotracheal tube was placed allowing a conscious patient to be intubated and subsequently anaesthetized when the airway was secure. This is fraught with considerable risk to the airway and requires the careful judgement of an experienced anaesthetist if undertaken. Although most ofthe swelling in Ludwig's angina is produced by inflammatory oedema there may be a collection of pus. This must be drained. Before the development of antibiotics the mainstay of treatment was by surgical decompression (Taffel & Harvey 1942) achieved by submandibular incision, sometimes in the form of a horseshoe creating access to both sides. This technique affords division ofthe deep cervical fascia, mylohyoid muscle and the anterior belly of the digastric allowing the tongue and floor of mouth to sink down. The resulting wound was packed lightly with vaseline gauze and allowed to heal by secondary intention. As well as preserving the airway this decreased the chance of infection spreading deeper into the neck and chest. Since this time the role of surgical decompression has lessened (Johnson et al. 1963), however, surgical exploration to ensure drainage of frank pus is still necessary and may be achieved by standard bilateral submandibular incisions. Corrugated rubber drains allow pus and the extensive inflammatory exudate to drain without blocking. They contribute to decompression and eventually enables the tongue to be returned inside the oral cavity. High blood levels of appropriate bactericidal antibiotics are valuable. Benzyl penicillin shows good activity against streptococci but it is likely to be inactivated by penicillinaseproducing staphylococci, Cefuroxime is a 'second generation' cephalosporin and is less

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