ORIGINAL ARTICLE Ludwig's Angina

African Journal of oral Health Volume 2 Numbers 1 & 2 2005: 16-23

ORIGINAL ARTICLE Ludwig’s Angina: An Analysis of Sixteen Cases in a Suburban Nigerian Tertiary Facility. Vincent Ugboko, Kizito Ndukwe, Fadekemi Oginni Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Obafemi Awolwo University, Ile-Ife, Nigeria.

ABSTRACT Objective: To document the prevalence and management of Ludwig’s angina in a suburban population. Methods: All consecutive cases of Ludwig’s angina seen and managed at the Obafemi Awolowo University Teaching Hospital, Ile-Ife, Nigeria between 1988 and 2002. Results: There were 16 cases with 10 males and 6 females whose ages ranged from 8 to 75 years; mean+ SD; 43.8 + 19.9 years. Symptom duration ranged from one day to three weeks; mean + SD; 6.4 + 4.9 days of the onset of illness. Odontogenic infection was the commonest etiologic factor in 12 cases (75%) and 4 of these were attributed to post dental extraction sepsis. Seven patients had underlying disease such as diabetes mellitus, lobar pneumonia, severe anaemia and mental retardation. The commonest was diabetes mellitus in 4 cases. Microbiological investigations showed a polymicrobial nature of the infection with Staphylococcus aureus (6 cases) out of 11 patients that had the examination. Treatment involved high doses of broad-spectrum parenteral antibiotics, immediate surgical drainage under local and general anaesthesia in 14 (75%) and single patients respectively. This was in addition to extraction of involved tooth/teeth where applicable. The complications recorded in 5 cases (31.3%) were septicaemia, mediastinitis, empyema thoracic, necrotising fasciitis, laryngeal spasm and renal failure. Mortality occurred in 4 cases (25%). Conclusions: Ludwig’s angina is a serious disease and late presentation remains a typical feature. Associated co-morbid condition is common. Prompt clinical evaluation and definitive care will considerably improve its prognosis and reduce the high mortality associated with the condition Key words: Ludwig’s Angina, Suburban Population, Tertiary Facility.

INTRODUCTION the floor of the mouth and Ludwig’s angina is a potentially life submandibular regions bilaterally and 1 threatening diffuse cellulitis involving causing progressive airway obstruction . The Stuttgart physician, Wilhelm Correspondence: Dr. VI Ugboko Frederick von Ludwig, first described Department of Oral and Maxillofacial Surgery this condition in 1836 as a nearly always Faculty of Dentistry, Obafemi Awolwo fatal facial space infection. University, Ile-Ife, Nigeria. E-mail: [email protected], Tel.: +234 36 233231 Fax: +448701349583 Typically, Ludwig’s angina is characterized by fever, malaise, Http://www.ajoh.org 1 6 ©African Journal of Oral Health

Ludwig’s Angina in a Suburban Facility – Ugboko et al African Journal of oral Health Volume 2 Numbers 1 & 2 2005: 16-23

and December 2002 constitute the dyspnoea, dysphagia as well as a brawny sample population for this study. hard tender swelling of the floor of the mouth and neck2-4. In most instances it Data were collected on presentation develops as a complication of an using a questionnaire proforma designed odontogenic infection usually from the to accommodate patients’ demographics, second and third molars5, 6. However, it cause and duration of infection, clinical may complicate cases of submandibular features and diagnosis. In addition, the gland sialadenitis and sialolithiasis, patients were made to undertake the tongue base lymphangioma, and tongue following laboratory investigations such piercing7-9 . as full blood count, electrolytes and urea, microbiology, culture and sensitivity The microbiology of Ludwig’s angina is pattern of pus aspirate, skull and chest polymicrobial and includes many gram- radiographs where indicated. Four cases positive and negative aerobic/anaerobic that were known diabetics had their organisms, but commonly isolated are blood sugar levels regularly monitored streptococcal spp, staphylococcus in conjunction with physician. Other aureus, prevotella spp and information obtained included treatment porphyromonas spp3, 10. given, outcome and complications.

Treatment invariably consists of Data were then tabulated and analyzed securing the airway where necessary, using simple frequencies and descriptive aggressive broad-spectrum antimicrobial statistics. therapy, and surgical decompression of the facial planes with removal of source RESULTS of infection3, 4, 6, 11. Over the study period, 16 patients presented with clinical features This paper is an analysis of sixteen cases consistent with Ludwig’s angina. There of Ludwig’s angina seen and managed in were 10 males (62.5%) and 6 females a suburban Nigerian Tertiary Facility. (37.5%) whose ages ranged from 8 to 75 years (mean+ SD; 43.8 + 19.9 years, PATIENTS AND METHODS median 35 years). Symptoms and signs Sixteen consecutive cases of Ludwig’s duration ranged from one day to 3 angina seen and managed by the weeks; mean + SD; 6.4 + 4.9 days Maxillofacial Unit of the Obafemi (Table1) with associated progressive Awolowo University Teaching Hospital, difficulty in breathing in most cases. Ile-Ife, Nigeria between January 1988

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Ludwig’s Angina in a Suburban Facility – Ugboko et al African Journal of oral Health Volume 2 Numbers 1 & 2 2005: 16-23

Table 1: Patients’ demographics, etiological factors and duration of disease at presentation. Patient Age (years) Gender Aetiology Duration (days) 1 60 Female Chronic periodontitis 4 2 27 male unknown 5 3 30 female chronic periodontitis 6 4 27 female unknown 1 5 65 male post extraction sepsis 7 6 25 female post extraction sepsis 7 7 35 male trauma 4 8 22 male apical periodontitis 3 9 55 male apical periodontitis 5 10 8 male trauma 5 11 60 female apical periodontitis 14 12 68 male chronic periodontitis 21 13 75 male post extraction sepsis 4 14 35 male pericoronitis 7 15 58 male post extraction sepsis 2 16 50 female chronic periodontitis 7

Table 2: Underlying systemic illness, treatment regime, complications and outcome in 16 cases of Ludwig’s angina. Patient Underlying Complications Outcome illness Treatment 1 Nil Surgical decompression/antibiotics Nil Alive 2 Nil Surgical decompression/antibiotics Nil Alive 3 Nil Surgical decompression/antibiotics Nil Alive 4 Severe anaemia Surgical decompression/antibiotics Nil Alive in pregnancy 5 Lobar Surgical decompression/antibiotics Nil Alive pneumonia 6 Mentally Surgical decompression/antibiotics Nil Alive retarded 7 Diabetes Surgical decompression/antibiotics Nil Alive mellitus 8 Nil Surgical decompression/antibiotics Nil Alive 9 Diabetes Surgical decompression/antibiotics Septicaemia, Died mellitus & mediastinitis hypertension 10 Nil Surgical decompression/antibiotics Nil Alive 11 Nil Surgical decompression/antibiotics Nil Alive 12 Nil Nil Laryngeal spasm, Died asphyxia, cardiac arrest 13 Diabetes Surgical decompression/antibiotics Mediastinitis, empyema Died mellitus thoracic & renal failure 14 Nil Surgical decompression/antibiotics Necrotising fasciitis Alive 15 Diabetes Surgical decompression/antibiotics Nil Alive mellitus Surgical decompression/antibiotics 16 Nil Laryngeal spasm, Died asphyxia, cardiac arrest

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Ludwig’s Angina in a Suburban Facility – Ugboko et al African Journal of oral Health Volume 2 Numbers 1 & 2 2005: 16-23

Odontogenic infection was the count also ranged from 8 to 15,000 X commonest aetiologic factor observed in 107/L while the electrolytes and urea 12 cases (75%), trauma was responsible levels were within the normal limits. for 2 (12.5%) while in the remaining 2 None was positive for the human patients (12.5%) the cause could not be immunodeficiency virus. The results of determined. Of those with odontogenic microbiology, culture and sensitivity origin, 4 (25%) were due to post dental tests from pus swabs in 11 patients extraction sepsis. At presentation, 14 (68.8%) revealed Staphylococcus aureus patients (87.5%) were pale, dehydrated (6 cases), haemolytic streptococcus (3 cases), Klebsiella pneumonia (2 cases) Fig. 1: Shows front view of patient 15 and one each of Pseudomonas at presentation with “croaking toad” aeruginosa, Proteus mirabilis, appearance. Echerichia coli, Prevotelaa intermedia and Citrobacter freundi. In three patients the culture yielded “no growth”, while anaerobic culture was carried out in only one case which yielded Citrobacter freundi and Prevotela denticola. The facility for routine culture of anaerobes is not available in our centre.

Fifteen patients (93.8%) were subjected to surgical decompression using interrupted submandibular and submental skin incisions, followed by blunt dissection of the fascial planes and and toxic looking with bilateral tender subsequent insertion of corrugated and brawny hard swelling involving the rubber drains. These remained in place submandibular, submental, and from between twenty four to seventy two sublingual spaces. Figure 1 shows a hours when there was virtually no typical case of Ludwig’s angina. There further discharge from the incision sites. was associated respiratory difficulty due The drainage was to relief the airway to gradual progression of the and this proved quite satisfactory for the inflammatory lesion to the neck in 11 13 patients who presented with airway cases (68.8%). compromise. Fourteen cases were carried out under local (infiltration) Seven patients (43.8%) showed clinical anaesthesia, while general anaesthesia evidence of underlying systemic illness. with orotracheal intubation was used in These were diabetes mellitus 25% (4 one patient. With regards to the patients cases) and 1 case (6.3%) each of managed under local anaesthesia, airway bilateral lobar pneumonia, severe anemia access was maintained using a Guedel’s in pregnancy and mental retardation. tube while oxygen was delivered (Table 2). through a facemask. None of the patients With exception of the diabetic patients, in this series had tracheostomy. Nine others had packed cell values ranging patients had removal of the offending from 11 to 23%, the white blood cell tooth/teeth and the incision sites were

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Ludwig’s Angina in a Suburban Facility – Ugboko et al African Journal of oral Health Volume 2 Numbers 1 & 2 2005: 16-23 closed following complete resolution of children3, 4, 7, 14. Most reports have been the infective process silent on the duration of symptoms before presentation at the hospital. It is Parenteral antibiotics, analgesics and expected that patients will present early multivitamin supplements were because Ludwig’s angina is a rapidly administered in all cases and the progressive disease. However, we antimicrobial regimen was a observed that over eighty percent of the combination of benzyl penicillin, patients in this series presented after the gentamycin and metronidazole. Where third day of the onset of their illness. appropriate, the antibiotics were This delay in presentation could be modified as dictated by the results of ascribed to distance from the rural sensitivity tests. The seven cases with referring centers, self-medication and associated systemic disease were abuse of antibiotics, ignorance and managed in conjunction with the patronage of unorthodox medical physicians and obstetrician. practitioners.

Septicaemia, mediastinitis, empyema Ludwig’s angina can arise from various thoracic, necrotising fasciitis, laryngeal sources such as odontogenic infection5, 6, spasm and renal failure were the or complicated cases of submandibular complications recorded in 5 cases. These gland sialadenitis and sialolithiasis, accounted for the death of 4 patients. Of tongue base lymphangioma, and tongue the dead, 2 had uncontrolled diabetes piercing7-9, 13, but several studies support mellitus in addition to Septicaemia, our finding that there is usually a dental mediastinitis and renal failure while the focus of infection5, 6, 12, 16. Details of the third case expired at presentation from pathway of spread have been well respiratory obstruction due to laryngeal documented4, 17. In previous reports14, 18, spasm during clinical examination in the 19 the role of underlying disease, emergency room. The fourth patient also particularly diabetes mellitus in the died from laryngeal spasm during aetiopathogenesis of severe orofacial extubation under general anaesthesia. infections was highlighted. Despite the fact that underlying systemic disease DISCUSSION was elicited in some of our cases (7/16), Ludwig’s angina is a rare condition and the literature is inconclusive as to this has been ascribed to the introduction whether systemic illness predisposes to of antibiotics several years ago5 and Ludwig’s angina. While sickle cell improved standards in dental practice4. anaemia, chronic alcoholism and This may also be responsible for the pulmonary infection were reported by 2 17 number seen during the period of the Odusanya and Hought et al , none of 4, study. There is no consensus of opinion the patients reported by other workers 10, 12 from previous investigations on the had associated systemic illness. demographic pattern of Ludwig’s angina However, what appears certain is that probably because of its uncommon such patients are immuno-compromised occurrence and the fact that majority of and should be thoroughly evaluated and studies were case reports7, 10, 12, 13. monitored, as they are readily However our findings are consistent susceptible to life-threatening with earlier reports showing male adults complications. The role of microbiology, preponderance with sporadic incidents in culture and sensitivity tests in the management of orofacial infections Http://www.ajoh.org 2 0 ©African Journal of Oral Health

Ludwig’s Angina in a Suburban Facility – Ugboko et al African Journal of oral Health Volume 2 Numbers 1 & 2 2005: 16-23 cannot be overemphasized. Overall, the drugs were appropriately modified isolates in the present study are depending on the outcome of sensitivity consistent with findings in previous tests. reports4, 13, 18, 19. However, inability to culture for anaerobes makes our Life threatening complications such as investigation incomplete and may be respiratory obstruction, mediastinitis, responsible for the negative routine pleural empyema, pericarditis, culture. pericardial tamponade are often associated with Ludwig’s angina6, 12, 13. The treatment of Ludwig’s angina This is in conformity with our study and consists of airway maintenance, surgical in conjunction with other factors drainage and broad-spectrum parenteral accounted for the fatalities we recorded. antibiotics4, 7, 10, 13. When there is airway Research has also shown that Ludwig’s compromise, airway management is angina has a mortality rate of 8-10%6, 16, probably the most important aspect of 23 and this occurs most often due to immediate care and should not be hypoxia or asphyxia rather than delayed6, 13, 20. Miller et al21 stressed the overwhelming sepsis12 which this study importance of airway management and has substantiated. The relatively higher recommended early contrast-enhanced rate in the present study can be attributed computerized tomographic (CECT) to late presentation, presence of imaging, as clinical examination alone uncontrolled underlying disease has a sensitivity of 55%. Neff et al20 especially diabetes mellitus, and recommended awake-fibreoptic economic constraints with inability to intubation to secure the airway if procure more effective prescribed computerized tomographic (CT) scan antibiotics. showed significant airway deviation or narrowing. In the face of limited Our findings suggest that inspite of the anaesthetic manpower and equipment, severity of this condition, late we relied on insertion of the Guedel’s presentation remains a typical feature airway and facemask for amongst the suburban populace but access/maintenance of the airways and prompt and thorough clinical evaluation supply of oxygen respectively. Prompt and definitive care will considerably incision and drainage undertaken in all improve the prognosis. While the need the cases available for treatment to secure the airway remains paramount, facilitated decompression of the facial local anaesthesia could be used safely spaces and also provided immediate and and it’s quite effective. progressive relief to the airway. In contrast with what obtained in other Acknowledgement: The authors wish to reports22-24, none of our cases had express their appreciation to Mr. Joe tracheostomy. Oyebanji of the Medical Illustrations Unit for his assistance with the The empirical antibiotic regimen in this photographic illustrations. series was combination chemotherapy involving benzyl penicillin, gentamycin and metronidazole and this was REFERENCES predicated on past clinical experience4, 1. Murphy SC. The person behind 18, 19 and knowledge of the usual the eponym: Wilhelm Frederick prevailing organisms. However, the Http://www.ajoh.org 2 1 ©African Journal of Oral Health

Ludwig’s Angina in a Suburban Facility – Ugboko et al African Journal of oral Health Volume 2 Numbers 1 & 2 2005: 16-23

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