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ORAL & MAXILOFACIAL SURGERY: CASE REPORT: Chronic Diffuse Sclerosing Osteomyelitis of the Mandible in a Young Patient; ORAL & MAXILOFACIAL SURGERY

July 1, 2000
by Bohdan Kryshtalskyj, BSc, DDS, MRCD(C), FICD and Simon Weinberg,


Chronic diffuse sclerosing osteomyelitis of the jaws is believed to be an inflammatory response to microorganisms of low virulence. Although carious, non-vital teeth have been implicated in the etiology and progression of this disease, chronic periodontal disease appears to provide a more common portal of entry for bacteria.

Clinical Features

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Although this condition may affect any age, sex or race, it tends to occur more often in middle aged Afro-American females. Typically, the disease follows a chronic protracted course with acute exacerbations of pain and swelling. The cortical bone may show significant expansion.

Radiographically, the process is diffuse, classically affecting a large part of the jaw. The lesion is poorly defined, consisting of early radioluscent zones in association with sclerotic masses, sclerosis tends to predominate as the condition progresses and periosteal thickening may also be observed.

CASE REPORT

A 14-year-old Philippine girl was referred to the oral and maxillofacial surgery service for an investigation and treatment of a gross left facial asymmetry. Her history indicated that in February of 1993 she had a severe dental infection. An incision and drainage was performed, followed by the removal of a deciduous tooth. Since the procedure, she developed a severe infection that drained through an extra-oral fistula under the left ramus of the mandible as well as the posterior border. She was treated with oral Amoxicillin for one month.

The fistula at the angle and posterior border of the mandible had resolved, but she had developed a continuing enlargement of the left ramus and body of the mandible. When she was seen in July of 1994, there was significant left facial asymmetry and a “hard” non fluctuant swelling of the left face. There was a 1.5 cm diameter fistula at the posterior aspect of ramus and 2.5 cm scar at the angle of the mandible. (Figures 1 & 2)

A CAT Scan was ordered and this showed that the left side of the mandible was markedly abnormal from the region of the angle to the mandibular condyle. The mandible in this region was markedly expanded and thickened in a somewhat irregular fashion, however, no cortical destruction was apparent and no soft tissue mass was identified. The abnormal section of the mandible extended to the region of the left molar. Enlargement of the left mandibular ramus, displaced the overlying soft tissue structures including the parotid gland laterally. The parotid gland itself appeared unremarkable, and there were no features suggesting malignancy. (Figures 3 to 5).

Additional films in the form of panoramic, lateral oblique view of mandible, parma view, PA skull, occlusal skull were ordered. These revealed the presence of a mixed radioluscent and radiopaque lesion in the left mandible from the second molar to involve all of the ramus, coronoid process, condylar neck and lateral aspect of condyle. There was a profound periosteal response, which was the greatest on the lateral aspect of the ramus of the mandible. There were multiple sequestrae within the mandibular ramus associated with new periosteal bone formation. After reviewing the CAT Scan and the films above, the diagnosis of chronic diffuse sclerosing osteomyelitis of the mandible seemed most likely.

Antibiotic therapy was begun consisting of: Pen VK 600 mg, po, qid elixir and Flagyl 250 mg, po, tid for the next six months.

Medical Treatment

The patient was referred to The Toronto General Hospital Hyperbaric Oxygen Facility. She had 20 preoperative treatments of hyperbaric oxygen prior to surgery. The dives were done at 2.5 atmospheres for 90 minutes Monday to Friday. Augmentation of the local host defenses and osteoclast remodeling required an extended course of hyperbaric oxygen post operatively and the recommended suggestion was 20 postoperative dives at which time a repeat CAT Scan and plain films would guide the clinical course and further treatment options would be initiated if necessary.

The hyperbaric oxygen dives would be in conjunction with prolonged antimicrobial treatment using Penicillin and Flagyl.

Surgery

Surgery was performed after 20 pre-operative treatments with hyperbaric oxygen. Utilizing a left submandibular Risdon approach, surgical exploration and corticotomy with sequestrectomy of the lateral aspect of ramus, condyle and anterior aspect of the body of mandible were completed. Biopsies of bone were submitted for aerobic cultures and sensitivity tests and gram stains. The fistulae were excised and surgical revision was performed.

The surgical findings revealed that after hyperbaric oxygen treatment and intensive antimicrobial therapy the bone appeared well vascularized with no evidence of active osteomyelitis. (Figures 6 to 8)

Report of Biopsies and Culture and Sensitivity Tests

The surgical specimen consisted of small pieces of bone with an aggregate diameter of 1.5 cm. The specimens were decalcified. In addition, a piece of bone measuring 1.5 cm in diameter was received directly from the operating room and immediately transferred to the microbiology laboratory for cultures and sensitivity tests, aerobic and anaerobic with gram staining; soft tissue was also submitted. The soft tissues included a small quantity of normal salivary gland tissue as well as lymph nodes exhibiting non specific chronic lymphadenitis. There was also an area in the bone which appeared to be necrotic. The intertrabecular space in that area was fibrotic. The findings were consistent with a sequestrum, possibly in the area of old osteomyelitis. The final diagnosis according to the above specimen was consistent with sequestrum, bone left mandibular ramus non specific chronic lymphadenitis, lymph nodes left mandible. The aerobic and anaerobic tissue cultures and sensitivities did not reveal any flora.

The surgical findings, showed well vascularized bone with no evidence of active osteomyelitis. Following the surgical treatment this patient underwent another 20 treatments with hyperbaric oxygen, and subsequent imaging of her mandible with a panorex and CAT Scan. These were compared to the films ordered 6 months previously. (Figures 9 to 11) In comparison the films showed:

– An overall reduction in the amount of periosteal new bone formation of the left mandible

– Considerable amount of bone remodeling into a normal but wide ramus

– No evidence of active inflammatory infection

– No evidence for the presence of sequestra

– The radiographic images were characteristic of healing after osteomyelitis.

DISCUSSION

The etiology of chronic diffuse sclerosing osteomyelitis remained obscure until 1994. Cultures usually came back as “negative” or “normal oral flora.” The reason for this may have been: inadequate specimens, delays in transportation of the specimens, or inadequate/ineffective culture techniques. In his timely article in 1994, Marx isolated two microorganisms responsible for this pathogenic infective entity, Actinomyces and Arachnia. These are slow growing filamentous Gram positive organisms that coalesce to form mycelia like colonies, which look like sulfur granules in clinical form. These organisms are facultative anaerobes. He has also isolated Eikenella corrodens. This organism is a Gram negative anaerobic coccus found in 50% of the cultures with actinomyces. Because of his findings, Marx recommended several new culture criteria:

– Culture the whole biopsy specimen rather then swabs.

– Use tissue grinding in thioglycolate broth with a SAGE 3500 tissue grinder

– Discontinue antibiotics one month before the biopsy

– Transport of cultures within a very short time period (15min) and place the innoculum into an anaerobic transport medium

– Slow growing organisms must hold anaerobic plates for seven days, therefore if a culture is 5 days or less you usually will get a normal flora or a negative result

In this case the only exception was the failure to discontinue the antibiotics one month before the surgery because of the severity of the osteomyelitis.

The Significance of Hyperberic Oxygen

Hyperberic oxygen cause
s the following tissue changes to occur:

– It reverses the hypovascular, hypocellular, hypoxic and fibrotic nature of the disease process within the bone and tissue.

– It enhances the neutrophil and macrophage activity, which are essential in fighting bacterial infections.

– It induces neovascularization through fibroblastic proliferation and collagen formation.

– It is bactericidal to anaerobes.

– It increases the concentration of antibiotic to damaged tissue.

– It replaces and encourages replacement of devitalized bone with healthy tissue and the formation of sequestra is enhanced.

However there are several contraindications to hyperberic oxygen therapy. The patient must be assessed prior to the prescribed treatment. Contraindications include: optic neuritis, neoplasia, active disease states such as COPD (Chronic Obstructive Pulmonary Disease) or seizure activity, claustrophobia, viral diseases and immunosupression. Other benefits of hyperberic oxygen is that it decreases pain and trismus, it closes fistulated wounds, and stimulates the proliferation of granulation tissue and healing.

A CT Scan and monthly follow -up showed a significant decrease in reactive bone formation. She has a symmetrical face and normal quality of life (Fig. 12).

Oral Health welcomes this original article. References are available upon request.