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Abstract
Osteomyelitis of the maxillofacial skeleton is a rare condition in recent times. The combination of antibiotic therapy and surgical debridement is effective in the treatment of chronic suppurative osteomyelitis (CSO). This case report describes the successful surgical treatment of CSO of the mandible in an 18-year-old adult. Treatment included a pre-surgical course of antibiotics followed by sequestrectomy, resection of the coronoid process and removal of the pathologically fractured condylar process of the left side of the mandible. On post-operative clinical review at 1 week, the extra oral draining sinus healed with improved mouth opening.
Osteomyelitis is the inflammation of bone and bone marrow that develops in the jaws after a chronic odontogenic infection or a variety of other reasons. [1] Advances in the field of anesthesia, antibiotic therapy, preventive and restorative dentistry, as well as the availability of component medical and dental care have reduced the incidence of the disease. [2] Chronic osteomyelitis may show a suppurative course with abscess or fistula formation and sequestration at some stage. [3]
Several reports have concluded that chronic suppurative osteomyelitis (CSO) can be treated successfully by a combination of antimicrobial therapy with surgery, either sequestrectomy or decortication of the affected bone. The aim of surgery is to eliminate all of the infected and necrotic bony tissue and, if incomplete, surgical debridement may lead to persistence of the osteomyelitis.

Case Report
An 18-year-old adult male was referred to the Department of Oral and Maxillofacial Surgery, M.R. Ambedkar Dental College and Hospital, with a 3-month history of discharging pus from a cutaneous sinus present on the left inferior border of the mandible[Figure 1]. His past dental history revealed that he underwent extraction of mandibular posterior tooth by dentist 1 year back. Painful swelling with discharging sinus occurred in the submandibular region after 2 months of extraction, which was treated by incision and drainage by the general surgeon. Six months later, pus discharge recurred, which was treated a second time by incision and drainage by a general surgeon, and this did not subside. He had no known allergies and denied any tobacco or alcohol use. On examination, a discharging extraoral sinus was present 2 cm below the lower border of the mandible [Figure 1], with tender left submandibular lymphnode. Two extraoral scars were observed, one on the skin 1 cm above the left side of the lower border of the mandibular region and another large scar on the left neck region postero-inferior to the left angle of the mandible, indicating poorly planned surgical intervention by the general surgeon [Figure 1]. There was limitation of mouth opening without any paraesthesia of the lower lip and mental area. On intra-oral examination, the patient had a reasonably well-maintained dentition.
Recurrentchronic0 Figure 1: Extraoral photograph showing draining sinus and two unesthetic scars resulting from previous interventions by a general surgeon

On radiological investigation, the panoramic view showed a localized mottled area of radio-opacity at the sigmoid notch, which was ovoid in shape and measured 20 mm at its greatest diameter. The radiographs suggested that there might be the presence of sequestrum [Figure 2]. There was a radiolucent line at the subcondylar region, suggestive of pathological fractures of condyle [Figure 2]. Based on clinical and radiographic findings, the patient was diagnosed as CSO of the condylar region. Treatment planned for the patient was pre-operative antibiotics for 1 week and sequestrectomy and removal of the pathologically fractured condyle followed by post-operative antibiotics for 1 week. Under general anesthesia, intra-oral incision was placed posterior to the third molar region, extending superiorly along the anterior border of the ramus. With careful reflection of the mucoperiosteum, it was surprising to see the discolored coronoid process showing the features of compromised blood supply and infected coronoid process. On table, we planned for coronoidectomy and sequestrectomy. Standard pre-auricular incision was placed with careful dissection of various layers to expose the temporomandibular joint and the pathologically fractured condyle was removed. Post-operative antibiotics were continued for 1 week.
Recurrentchronic1 Figure 2: Panoramic view showing a radio-opaque lesion in the sigmoid notch area (sequestrum) and radiolucent fracture line in the condylar neck region

The histopathological report revealed chronic inflammatory cell infiltration, with areas of resorption of bony trabacula. These findings, in combination with the clinical picture and radiological features, were consistent with CSO. The draining sinus healed well with improved mouth opening at 1 week post-operatively. The pre-auricular region also healed well without scar [Figure 3]. Three months and 6 months after the original surgery, repeat radiographs were taken. There was no clinical or radiological evidence of residual infection[Figure 4] and [Figure 5].
Recurrentchronic2 Figure 3: Post-operative photograph showing well-healed pre-auricular region without scar

Recurrentchronic3 Figure 4: Axial computed tomography scan of the patient

Recurrentchronic4 Figure 5: Seven-month post-operative orthophantamograph


Discussion
Osteomyelitis is an inflammatory condition of the bone that involves the medullary cavity and has a tendency to progress along this space and involve the adjacent cortex, periosteum and soft tissue. [4] It is more common in the mandible than in the maxilla because of the dense, poorly vascularized cortical plates and the single blood supply from the inferior alveolar neurovascular bundle. [4] The primary cause of the chronic osteomyelitis is usually microbiologic and results from an odontogenic infection, post-extraction complications, inadequate removal of necrotic bone, early termination of antibiotic therapy, inappropriate selection of antibiotics, diagnostic failure, trauma, inadequate treatment for fracture or irradiation to the mandible. [1] The most common bacteriologic results reported to the treating clinicians were mixed oral flora or mixed anaerobic flora. [2] The distribution of osteomyelitis in the jaws dominated by cases that occurred in the mandible, with the highest frequency found in the angle and the body regions. [2] In chronic secondary osteomyelitis, the clinical findings usually are limited to fistulas, induration of soft tissue and thickened or wooden character to the affected area, with pain and tenderness on palpation. In cases of recurrence, symptoms often occurred immediately adjacent to the decorticate area. [5] Culture, bone biopsy, conventional radiography, radioisotope bone scanning, laser Doppler flowmetry, computerized tomography and magnetic resonance imaging are used to diagnose chronic osteomyelitis. [1]

Management entailed a course of antibiotics in combination with surgical debridement. In CSO of the mandible, several authors recognize resistance to therapy as an infrequent but possible problem. Topazian recommends to continue post-surgical treatment for 2-4 months after the resolution of the symptoms where as Bartkowski et al. use intravenous therapy for 10-24 days. This is consistent with the published protocols of Van Merkesteyn et al. It has been suggested that antibiotic therapy combined with surgical intervention is effective in the treatment of CSO. [6] Some reports have also advocated the use of hyperbaric oxygen in the treatment of this condition, especially in the irradiated mandible. In the present case, the patient was prescribed a course of antibiotics, which, in combination with surgical intervention, was successful.

Conclusion
Proper diagnosis and treatment planning is of utmost importance to cure any disease. In this case, improper diagnosis and treatment plan resulted in the recurrence of lesion with unesthetic scars in a young adult. Considering clinical presentation and course of the disease with successive previous treatment failure, our emphasis was given to the source of infection, which was successfully treated by surgery and antibiotic course.
 
References
1. Kim SG, Jang HS. Treatment of chronic osteomyelitis in korea. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;92:394-8. 
2. Koorbusch GF, Fotos P, Goll KT. Retrospective assessment of osteomyelitis etiology, demographics, risk factors, and management in 35 cases. Oral Surg Oral Med Oral Pathol 1992;74:149-54.
3. Baltensperger M, Gr?tz K, Bruder E, Lebeda R, Makek M, Eyrich G. Is primary chronic osteomyelitis a uniform disease? Proposal of a classification based on a retrospective analysis of patients treated in the past 30 years J Craniomaxillofac Surg 2004;32:43-50. 
4. Fullmer JM, Scarfe WC, Kushner GM, Alpert B, Farman AG. Cone beam computed tomographic findings in refractory chronic suppurative osteomyelitis of the mandible. Br J Oral Maxillofac Surg 2007;45:364-71. 
5. Montonen M, Iizuka T, Hallikainen D, Lindqvist C. Decortication in the treatment of diffuse sclerosing osteomyelitis of the mandible: Retrospective analysis of 40 cases between 1969 and 1990. Oral Surg Oral Med Oral Pathol 1993;75:5-11. 
6. Van Merkesteyn JP, Groot RH, van den Akker HP, Bakker DJ, Borgmeijer-Hoelen AM. Treatment of chronic suppurative osteomyelitis of the mandible. Int J Oral Maxillofac Surg 1997;26:450-4. 

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Dolphine Oda, BDS, MSc
University of Washington Oral Pathology 

Case and images contributed by: Drs. Mark Egbert, Rafael Alcalde & Tarja Kaakko Seattle Children’s Hospital & University of Washington

History

An 11-year old Hispanic female presents to the University of Washington Pediatric Dentistry clinic. There is a rapidly growing swelling in the right posterior mandible displacing teeth #s 30 and 31. It has been present or one month. No history of trauma.

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 Presented by
Dolphine Oda, BDS, MSc
Case and images contributed by: Dr. James Reed, Edmonds, Washington
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History of Present Illness
A 47-year-old Hispanic male was referred from the emergency department with left facial swelling, occasional oral bleeding, facial swelling for 5 days, bitter taste, and fevers of up to “99.0 F”. Symptoms and signs had been present for one month.

 

 

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History


Chief Complaint: This 74 year old female was referred because of complaint of progressive pain in the right face and limited jaw movement.

Medical History: Positive for breast cancer, arthritis, hypertension, and knee replacement; negative for blunt trauma to the jaw or face. No known allergies. Current medications included Zolpidem, HCTZ, Tramadol, and Prilosec. The patient slept 8 hours nightly and imbibed occasional caffeine.

Family Medical History: Heart disease, HBP, Cancer.

Review of Systems: Eye pain, GERD, headache, chewing difficulty right side.

Examination: Vitals: 165/90; Jaw Opening (ROM) 40mm with pain; maximum opening of 55 mm associated with right sided pain; crepitation bilateral L>R; occlusal contacts bilateral and stable; cranial nerves 2-12 within normal limits; myofascial: right mass and temporalis tender points; cervical within normal limits for her age; lateral capsular tenderness right TMJ

Intraoral: Probable geographic tongue; mild dental attrition

Radiography: See included CBCT images with specific emphasis on the right Temporomandibular joint.

Preliminary Impression: Arthromyalgia right TMJ

Final Diagnosis:
  1. OA Bilateral TMJ with loose body on left
  2. Masticatory Myalgia
  3. History of CA
      Treatment Plan: Plan diet changes; splint use pm's; symptomatic medications; topical anti-inflammatory medication

      This case is courtesy of Bradley Eli, DMD, MS
      Director, San Diego Headache and Facial Pain Center

      Assistant Professor, UCSD School of Medicine, Anes

      Dental School 1987 Temple University

      Post Doctoral education UCLA cert in Orofacial Pain, Masters in Oral Biology 1992

      By Steven Guttenberg, DDS, MD



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      Case presentation: Chief complaint/ Brief History: The patient is a 30 year-old left handed female who presented with the following complaints: 1) moderate pain in her left jaw joint 2) mild pain in her right jaw joint, 3) pain radiating from the back left side of her head, and 4) bite changes. She reports having been struck in the face seven years ago while she was attending a concert. She immediately had clicking and popping sound in her left jaw joint. In the last two years she started having pain in the left jaw joint. Her bite began to change during the last six months. She has seen by an Oral and Maxillofacial Surgeon who did arthrocentesis twice in her left jaw joint and has injected steroid medication recently. A report from a Medical MRI detailed a normal left temporomandibular joint with mild changes on the right.

      Extra-oral findings:
      Moderate tenderness to palpation of the left temporomandibular joint. Palpation of the left masticatory and cervical muscles revealed tenderness and multiple trigger points.


      Intra-oral findings: In the closed position she only contacts her left second molars. No other teeth occlude. Range of motion 50 mm intra-incisal with good lateral movement. Crepitus is noted in the left temporomandibular joint with movement. Working diagnosis Capsulitis of the Left Temporomandibular Joint, Severe Malocclusion, and Myofascial Pain Dysfunction.

      Imaging:
      The MRI report did not seem to correlate to the clinical findings. The patient was referred to the UCLA School of Dentistry Radiology Department. for Morita Cone-Beam Volumetric Tomography. The films were taken and reviewed by their Oral and Maxillofacial Radiologist, Sotirios Tetradis, DDS, PhD. A diagnosis of Osteoarthritic Changes of the left temporomandibular joint was made. This diagnosis correlated to the clinical findings. (See attachment of images)


      Differential Diagnosis: Traumatic Arthrits, Idiopathic Condylar resorption, R/O Autoimmune arthrits (Rheumatoid, Psoriatic arthritis, etc) Laboratory and clinical evaluation revealed no positive findings.

      Final Diagnois: Traumatic Osteoarthtis of the Left Temporomandibular Joint with bite changes.

      Brief Discussion: It is interesting to see the late development of pain and developing malocclusion after the initial trauma. There is some discussion in the literature of this being associated more frequently with females with estrogen dysregulation. One wonders if TNF reducing medications such as Enbrel would be of any benefit (if it were not for their potential side effects). Could such medication be placed in the joint directly? For now watching for occlusion changes on a balanced occlusal splint (worn part-time) to monitor changes and the use of non-steriodal anti-inflamatory medication as needed to reduce pain and swelling.This is included with a myofascial protocol to limit her facial pain. The patient has been told when her occlusion does not change and she is pain free for at least six months corrective orthodontic and possible orthognathic procedures will be considered with no guarantee that her bite will remain stable.

      Thanks to the UCLA School of Dentistry Radiology Department and Sotirios Tetradis, DDS, PhD Douglas R. Wall, DDS is in private practice in Long Beach, California He is a Visiting Lecturer and Part-Time faculty at the UCLA School of Dentistry Orofacial Pain and Sleep Medicine Residency as well as an Associate Professor and Part-time faculty at the USC School of Dentistry Orofacial Pain and Oral Medicine Residency

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      Panoramic x-ray
      This panoramic radiograph was presented to a radiologist for interpretation. According to the provider, at the time of presentation the patient had no significant history, signs, or symptoms.
      What and where is the structure of interest?

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      Dolphine Oda, BDS, MSc (Pathology)
      Dr. Tracy Johnson (Oral & Maxillofacial Surgery)

      History
      This 30-year-old female presented for wisdom tooth removal.  At examination of the patient it was noted that there was swelling in the region of the right posterior maxilla.  How long the lesion had been present was unknown.  
      Medical History
      Unremarkable
      Clinical and Radiographic Findings
      A large asymptomatic swelling was identified by the oral surgeon in the right posterior maxilla.
      It was confirmed by a panoramic radiograph which showed a well-demarcated mixed radiolucent/radiopaque lesion between teeth #s 3 and 4 (Figure 1).  It had expanded buccally and palatally and had extended superiorly into the right maxillary sinus. It was displacing teeth #s 3 and 4. The lesion was measured as 2 x 2 x 1.7 cm in size. The involved teeth were vital.
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      Case seen by Dolphine Oda, BDS, MSc; submitted by Wesley Hanson DDS MSD
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      Pre-operative picture of patient and CBCT scan showing his severe airway restriction
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      Ameloblastoma revealed in i-CAT® scan
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      Dolphine Oda, BDS, MSD
      Contributed by Drs. Mark Carlson, Galya Pirinjian
      Tacoma Oral Surgery and University of Washington
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      History of Present Illness
      A 60-year-old white male who is evaluated severe and localized bone loss in the upper right posterior maxilla.
      Medical History

      The medical history is significant for hypertension and hypercholesterolemia. The patient has no known drug allergies. The patient had a CAT scan done in June 2003 for symptoms of pneumonia and possible sepsis (not shown). He also had CT of the head, chest, abdomen, and pelvis, in an attempt to diagnose the etiology of the pneumonia. The radiologist noticed an expansile intermediate-density mass in the right posterior maxilla, causing bony destruction and extending inferiorly into the maxillary sinus, measuring 2.5 X 2.1 cm in greatest dimensions.
      Clinical & Radiographic Findings
      The clinical examination showed an area of severe and localized bone loss in the right posterior maxilla simulating localized periodontitis which lead to the extraction of tooth #2. At extraction, the dentist noticed that the bony consistency was soft and spongy combined with extensive bone loss involving the posterior maxilla and the inferior and posterior portions of the maxillary sinus (Fig 1). The Oral Surgeon also noticed bony expansion in addition to the extensive destruction. The bony destruction at that point was measured to be approximately 3 ¼ x 2 ½ cm in greatest dimensions (Fig 1).

      Figure 1. Panoramic view of the presentation in Dec 03 demonstrating destructive radiolucency involving the right posterior second molar tooth, distal bone and the maxillary sinus.
      Incisional Biopsy
      Histologic examination revealed a solid and benign neoplasm of odontogenic epithelial origin made up of a combination of epithelial islands and cords (Fig 3, 4). The periphery of the islands is lined by columnar or cuboidal and palisaded epithelial cells (Fig 4) and the center of the islands is filled with cuboidal and stellate epithelial cells consistent with stellate reticulum. The epithelial cords are long and are forming a network of two-layers of cuboidal and palisaded epithelial cells. These epithelial islands are suspended on a background of loose and vascular connective tissue.

      Figure 3. Low power (x100) histology shows odontogenic epithelial islands and cords. The periphery of the islands is lined by columnar or cuboidal and palisaded epithelial cells and the center of the islands is filled with cuboidal and stellate epithelial cells.
      Figure 4. Higher power (x200) histology shows epithelial islands and cords lined by columnar cells, some demonstrating reversed polarization.
      Differential Diagnosis
      Odontogenic Keratocyst
      Ameloblastoma
      Central giant cell granuloma
      Brown tumor of hyperparathyroidism
      Malignant salivary gland neoplasm
      Final Diagnosis
      Hint: This lesion is a slow-growing, persistent, and locally aggressive neoplasm of epithelial origin.
      http://mydental.uw.edu/oralpath/caseofthemonth/oct-04/diagnosis.htm
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      Case Presentation
      Juan F. Yepes DDS, MD, MPH, FDS RCSEd, DrPH
      Medical Institutional Review Board Vice-Chair
      University of Kentucky
      jfyepe2@email.uky.edu



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      A 45-year-old Hispanic male physician, a nonsmoker, presented with crippling, nonerosive, seropositive, symmetrical polyarticular arthritis that involved his wrists, hands, elbows, knees, and feet. Findings included an erythrocyte sedimentation rate (ESR) of 114 mm/h (normal, lower than 23 mm/h) and a weakly positive anti–cyclic citrullinated peptide antibody result. The patient had a history of migraine and tinnitus. He had a previous diagnosis of serous otitis media with middle ear and mastoid effusions on CT scanning; he responded initially to antibiotic and corticosteroid therapy.

      A diagnosis of rheumatoid arthritis (RA) was made, and the patient was treated with oral methotrexate(Drug information on methotrexate) and self-injected etanercept(Drug information on etanercept). His inflammatory arthritis with disabling musculoskeletal symptoms resolved. However, in spite of a rapid taper of low-dose prednisone(Drug information on prednisone) early in the disease course, the patient continued to self-prescribe various doses of corticosteroids for worsening headache and tinnitus; all symptoms were relieved.

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