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July 17, 2007

By James J. Sciubba, DMD, PhD

Case 2-2007
This is the case of a 69-year-old woman with a rapidly progressive postextraction complication.

Case Presentation

A 69-year-old woman developed a cellulitis over the right mandibular body 4 weeks after routine extraction of the mandibular second molar, which was deeply carious and associated with severe pain. The extraction was performed without difficulty under local anesthesia, with no immediate postoperative complications. Over the ensuing 2 weeks, a modestly tender swelling developed over the right lower third of the face over the extraction site, with pain along the mandibular buccal region. The swelling enlarged in association with mild trismus, a fetid odor, and a low-grade fever.

Medical History

The patient essentially had been well until approximately 10 years earlier, when she was diagnosed with a right neuroretinitis by her ophthalmologist. Shortly thereafter she developed a rheumatologic disorder of unknown etiology, with widespread joint pain and severe fatigue. In an effort to rule out systemic lupus erythematosus and vasculitic disorders such as Wegener’s granulomatosis, a laboratory workup was performed; results included a borderline-positive antinuclear antibody level, a nonspecific perinuclear anti-neutrophilic cytoplasmic antibody positivity pattern, and negative myeloperoxidase and proteinase 3 test results as measured by enzyme-linked immunosorbent assay. A suspected myocardial infarction was evaluated with no performance compromise or other abnormalities noted at the time of her initial cardiac workup.

Approximately 18 months prior to being seen by her dentist for right mandibular pain, she was treated with oral glucocorticosteroidsⁱ for her rheumatologic complaints, with a good response noted. Steroids were discontinued using a slow, tapering dosing schedule over a period of several weeks. Six months later, she was evaluated for elevated Mycoplasma levels on a titer test and for recurrent severe fatigue and joint pain, and was again treated with an oral steroid regimen. After this she was suspected of suffering from Sjögren’s syndrome and autoimmune thyroiditis. Serum inflammatory markers, including C-reactive protein and erythrocyte sedimentation rate, were elevated. An earlier echocardiogram had revealed an ejection fraction of 40% to 50%.

During the next several weeks a slow taper of the steroids was initiated. However, a recurrence of severe fatigue and joint pain required reinitiating the steroid dosage at 12.5 mg daily. Further medical workup for celiac sprue, Whipple’s disease, and Wegener’s granulomatosis was negative, as were results on enteroscopy.

At this point severe pain developed within the right mandible, where a nonrestorable second molar was noted by her dentist (Figure 1). A routine simple extraction of the nonrestorable tooth was performed uneventfully by an oral surgeon. Four days later she developed a right lower facial cellulitis, with a subsequent diagnosis of mandibular osteomyelitis. After the development of facial swelling and cellulitis, she was placed on a series of oral antibiotics and, soon thereafter, a brief course of intravenous antibiotics, including vancomycin and piperacillin/tazobactam. No improvement of the cellulitis was noted. A routine panoramic radiograph (Figure 2) demonstrated a mottled area of bone destruction at the extraction site that extended toward the inferior cortical margin of the mandible. She was subsequently hospitalized for management of the persistent oral infection and her general complaints of fatigue. An echocardiogram performed on admission revealed an ejection fraction of 20% to 25%, along with the presence of a large left pleural effusion. The origin of her left ventricular dysfunction was unclear, although the possibility that this was related to her underlying undiagnosed rheumatologic disease was considered. A routine complete blood cell count showed an elevated white blood cell count of 26.5.

The initial oral and head and neck examination in the hospital revealed a mild, diffuse, and slightly tender right facial swelling centered over the mandibular body, with an attendant degree of mild cutaneous erythema and trismus (Figure 3). Intraorally a gray-colored area of necrotic soft tissue was present along the right buccal aspect of the mandible, extending into the mucobuccal fold (Figure 4). The tissue was insensate, densely fibrous and did not bleed upon manipulation. In addition, and of note, the buccal cortical bone in the molar and premolar region was exposed in the absence of suppuration. The adjacent dentition was intact, firm, and without evidence of periodontal disease. The tooth socket at the recent extraction site was open, and the alveolar bone was exposed, with no sign of granulation tissue along the axial walls or at the base of the defect. A polymicrobial infection was suspected and later confirmed by culture, which identified aerobic, anaerobic, and facultative anaerobic bacteria. This prompted initial treatment with intravenous gatifloxacin and metronidazole, accompanied by parenteral hydration and analgesics. A subsequent bone scan of the mandible indicated findings consistent with osteomyelitis of bacterial origin.

Computed tomography revealed soft tissue swelling within the submandibular and retromandibular areas, with fluid accumulation in association with aggregates of gas within the soft tissues on both the lingual and buccal aspects of the mandible (Figure 5). Extensive cellulitis of the right neck was present, along with hypopharyngeal asymmetry. After the imaging studies, aggressive surgical debridement of the necrotic tissue was performed intraorally and in the anterior neck, followed by pathologic evaluation of the removed tissue.

Pathologic analysis of the submitted tissue demonstrated broad areas of tissue necrosis, bacterial overgrowth, separation of skeletal muscle bundles and nerve trunks, muscle necrosis, and a mixed inflammatory infiltrate (Figures 6 and 7). No fungal organisms were present on special fungal staining. In concert with the clinical, pathologic, and imaging findings, a diagnosis of necrotizing fasciitis was made.

At 17 days after admission, her condition continued to deteriorate in spite of aggressive antibiotic treatment and cardiac and pulmonary support. She developed bilateral pulmonary emboli with an associated sharp increase in leukocyte count, to 88,000/mm.³ She later became hypotensive, requiring vasopressors in the presence of persistent sepsis, and developed severe bradycardia. She developed pulseless electrical activity and did not respond to appropriate measures to reestablish a heart rate.

At autopsy the diagnosis of multiorgan failure was made secondary to cellulitis/sepsis originating in the right neck and jaw region. The dental infection was deemed to be the initiating event and its progression the cause of death, within the context of undefined rheumatologic disease that required significant and prolonged systemic immunosuppressive treatment.

Discussion

Necrotizing fasciitis is an uncommon, highly lethal, and rapidly developing disease, spreading polymicrobial infection often associated with group A streptococci and several other organisms, including Clostridium perfringens and Streptococcus pyogenes and the necrotoxins produced therein.¹ On occasion a single organism may produce some necrotizing infections, but in the current case the origin of the infectious process was odontogenic, which is typically polymicrobial or mixed in type. Such mixtures of organisms include aerobic, anaerobic, facultative anaerobic, and obligate anaerobic species.

Although this condition is rare, dental professionals often are the first to encounter it, as most cases of necrotizing fasciitis of the head and neck region are chiefly the result of an odontogenic infectious process, as in the case described here. The use of immunosuppressive drugs (glucocorticosteroids) to manage the ill-defined rheumatologic disease is likely to have contributed to the rapid progress of the infection, but a significant percentage of cases arise in patients who are otherwise well. The mortality rate associated with this disease in patients with attendant systemic conditions was 24.2% in 1 study, but ranges upward from 19.2%; previously healthy patients had a reported 9.3% mortality rate.² An assessment of the literature demonstrates a significantly higher percentage of fatal outcomes in patients with attendant systemic disease - particularly diabetes mellitus and alcoholism - than in patients who are otherwise healthy. Poor outcome is also associated with the time that elapses until aggressive surgical debridement, with patients who undergo this procedure within 24 hours having a significantly lower mortality rate (19.6%) than those who deferred surgery for more than 24 hours (50%).^2,3

Factors responsible for the initiation and progression of tissue damage, in addition to local microbial and host resistance factors, include factors produced by the invading microflora. These include potent necrotoxins and activation of the coagulation system, which in turn leads to formation of thrombi and tissue infarction. Heparinases produced at the infectious site further abet the procoagulative events. Elevations in local hydrostatic pressure further inhibit local vascular perfusion within the area.

Given the polymicrobial nature of the infection, this case is best considered a Type I necrotizing fasciitis. In contrast, where documented S. pyogenes (group A Streptococcus) is present, Type 2 necrotizing fasciitis is designated, and when clostridial species are isolated, clostridial myonecrosis (gas gangrene) is diagnosed.⁴

This severe life-threatening condition, when affecting the head and neck region, is most commonly associated with an odontogenic infection (apical, periodontal) as the seminal or initiating clinical event. This condition may be mistaken for a more typical odontogenic infection, as its early presentation may not be specific. In addition, the isolated organisms are predominantly β-hemolytic Streptococci, Staphylococcus, and Bacteroides spp., and common resident oral and periodontal pathogens. Diagnostic considerations include the presence of pronounced pain and systemic toxicity (leukocytosis, elevated C-reactive protein levels), the presence of gas within local soft tissues, and identification of tissue breakdown. Underlying immunosuppression and concurrent systemic disease further increase the risk of progression.

Management must be aimed at immediate surgery in an effort to reduce the risk of mortality. It is crucial to remember that dental infection is the main cause of this condition when it occurs in the head and neck area

Summary

An example of a common odontogenic infection requiring a routine dental extraction is presented within the broader context of an iatrogenically immunocompromised host having an ill-defined rheumatologic disease, with a fatal outcome secondary to a polymicrobial necrotizing infection of odontogenic origin. A broad area and high volume of tissue necrosis was found, with extension well beyond the mandibular site of origin. Clinicians must adequately evaluate immunocompromised patients with regard to potential systemic consequences prior to instituting ambulatory surgical procedures that would normally be considered routine. Finally, once the correct diagnosis is established, immediate and aggressive surgical debridement and supportive management are crucial.

References

1. Chapnick EK, Abter EI. Necrotizing soft tissue infections. Infect Dis Clin N Am. 1996;10:835-855.
2. Umeda M, Minamikawa T, Komatsubara H, et al. Necrotizing fasciitis caused by dental infection: a retrospective analysis of 9 cases and a review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2003;95;283-290.
3. Elliott DC, Kufera JA, Myers RA. Necrotizing soft tissue infections: risk factors for mortality and strategies for management. Ann Surg. 1996;224:672-683.
4. Urschel JD. Necrotizing soft tissue infection. Postgrad Med J. 1999;75:645-649.

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