Osteomyelitis Presenting in Two Patients: a Challenging Disease To

Osteomyelitis presenting IN BRIEF • Osteomyelitis should be considered as a differential diagnosis in patients PRACTICE complaining of chronic pain post-dental in two patients: a challenging extraction or injury. • A good clinical and patient history is required as clinical and radiographic signs disease to manage may present late. • Oral antibiotics appear to have minimal 1 2 3 impact as initial treatment. V. Patel, A. Harwood and M. McGurk • Cone beam CT may help conclude a diagnosis earlier.

Chronic osteomyelitis of the jaw is a rare entity in the healthy population of the developed world. It is normally associated with radiation and bisphosphonates ingestion and occurs in immunosuppressed individuals such as alcoholics or diabetics. Two cases are reported of chronic osteomyelitis in healthy individuals with no adverse medical conditions. The management of these cases are described.

INTRODUCTION the nomenclature discussed by Eyrich et injections of local anaesthesia. Her medi- Osteomyelitis can be defi ned as an infl am- al.,6 primary chronic osteomyelitis (PCO) cal history was non-contributory and she matory condition of the bone, which is defi ned as chronic non-suppurative had smoked approximately 20 cigarettes begins as an infection of the medullary osteomyelitis; when PCO occurs in chil- a day for the past fi ve years and did not cavity, rapidly involves the haversian dren and adolescents it is termed ‘Garré’s drink alcohol. systems, and extends to involve the peri- osteomyelitis’. This is in contrast to sec- On examination the extraction socket osteum of the affected area.1 It is a well ondary chronic osteomyelitis (SCO), which was red and infl amed indicative of local known entity in the historical literature is chronic osteomyelitis with suppuration, osteitis (dry socket). A four week course of where in the absence of antibiotics, com- abscess/fi stula formation, and sequestra- clindamycin was prescribed which delayed pound fractures of long bones frequently tion at some stage of the disease due to a the symptoms initially but recurred on ces- failed to heal. Such cases are no longer defi ned, infectious aetiology.7 sation of the medication in September 2008. part of modern medical experiences. The complaint was of intense uncontrolla- In the twenty-first century osteomy- Presentation ble pain and a sensation of ‘loose teeth’. On elitis presents as a sub-chronic condi- Acute osteomyelitis is characterised by examination the patient was apyrexial and tion and is more commonly associated a virulent infection with intense pain, intra-orally there were no signs of infection with debilitated, immunosuppressed or infl ammation, redness and can be life at the extraction site. A full blood profi le medically compromised2,3 patients and threatening due to its toxic effects. If including ESR and CRP were reported as the pattern of events does not pose a however, the bacteria are less virulent, the normal. A MRI scan demonstrated a blush diagnostic dilemma. symptoms can differ and mimic an acute within the bone marrow cavity indicative and prolonged alveolar osteitis making it of oedema but lacked evidence of extensive Classifi cation diffi cult to diagnose and treat. bone involvement. A bone scan report sug- Acute osteomyelitis (AO) compared to This paper outlines two examples of gested the possibility of osteomyelitis but chronic osteomyelitis is differentiated this condition arising from routine den- should be considered in conjunction with arbitrarily based on time: an acute proc- tal procedures, detailing their mode the MRI. A second more intense course ess occurs up to one month after the onset of presentation and the distinguishing of antimicrobial therapy was commenced of symptoms and the chronic process features indicative of the condition. with a mixture of IV and oral antibiotics occurs for longer than one month.4,5 Using (azithromycin, teicoplamin, co-amoxiclav, CASE REPORT clindamycin and metronidazole) continued Case 1 over four weeks. The patient responded to the treatment and became symptom free 1Oral and Maxillofacial Surgery, 2Dental Radiology, In June 2008 a 47-year-old female was for six months. 3*Department of Oral & Maxillofacial Surgery, Guy’s Hospital, Floor 23, Great Maze Pond, London, SE1 9RT referred to the Oral and Maxillofacial At this point she again complained of *Correspondence to: Professor Mark McGurk Department with pain and swelling follow- intense pain and general malaise. A Cone Email: [email protected] ing the extraction of a lower right second Beam CT (CBCT) demonstrated bony defects Refereed Paper molar (LR7) by her general dental practi- in the LR7/8 area compatible with chronic/ Accepted 30 April 2010 DOI: 10.1038/sj.bdj.2010.927 tioner (GDP) a month earlier. The extrac- recurrent osteomyelitis (Fig. 1). Further ©British Dental Journal 2010; 209: 393–396 tion proved diffi cult and required repeat imaging was available from the CBCT

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Fig. 1 Coronal slice of the CBCT scan showing the irregular loss of the buccal and Fig. 2 Reconstructed panoramic image from the CBCT scan illustrating the area of bone loss superior alveolar cortices (arrowed) associated with the extracted lower right second molar tooth (arrowed) showing the extent of the bone involve- a sinus was present on the lingual aspect ment in Figures 2 and 3. A biopsy of the of the right mandible. There was no par- bone was uninformative as was micro- aesthesia but the patient complained of biology which reported ‘there is a pres- her ‘teeth becoming loose’ although this ence of growth of mixed anaerobes, some could not be demonstrated clinically. Viridans streptococci and Actinomyces She was commenced on intravenous naeslandii. This growth could be com- clindamycin for two weeks followed by patible with normal oral fl ora though co-amoxiclav for a further four weeks to Actinomyces can cause chronic osteo- which she responded well. Subsequently myelitis.’ The patient was recommenced the infection recurred, but now the pain Fig. 3 Surface rendered 3D reconstruction from the CBCT scan showing the area of on Ceftriaxone IV and Metronidazole PO was in the left mandible, for the infection bone destruction (arrowed) in the lower for a further four weeks. Currently this had run through the marrow spaces to right quadrant patient is symptom free and under long the contralateral side of the jaw. A CBCT term review. reported ‘widespread perforation of the lingual plate consistent with sub-perio- Case 2 steal spread of infection from the origi- In April 2008 a 67-year-old female was nal intra-osseous injection site across the referred complaining of an intense pain midline to affect the left premolar region’ in her lower jaw. The condition had been (Fig. 4). Bone biopsies were compatible ongoing for almost four months. Medically with sclerosing low-grade chronic osteo- she was fi t and well. myelitis. A repeat CBCT one year later The history revealed that in January showed regeneration of the mandible and 2008 she attended her GDP in Norway improvement in comparison to the previ- for root canal treatment of a lower right ous CBCT (Fig. 5). Fig. 4 Axial cone beam CT slice through the fi rst molar (LR6). Treatment was preceded mandible: multiple dehiscences of the left by a lingually applied intra-osseous injec- DISCUSSION mandibular lingual cortex (arrowed) as a tion of local anaesthesia. The follow- Osteomyelitis of the jaw is a relatively result of infection tracking sub-periosteally from the lower right molar/premolar region ing day she developed pain and lingual uncommon inflammatory disease in swelling which was treated with antibi- developed countries.8 The aetiology is otics and analgesics but without resolu- unknown and theories include bacte- tion. She consulted a second endodontist rial infection (dental or bacteraemia who thought the pain was pulpitis in the from distant foci), vascular defi ciency adjacent tooth (LR5) and proceeded to a (localised endarteritis), autoimmune dis- second root canal treatment. The chronic ease7 or trauma.9 Conditions altering the pain persisted and a month after pres- vascularity of the bone such as radia- entation she developed swelling on the tion, malignancy, osteoporosis, osteo- lingual aspect of the mandible, in the LR5 petrosis, and Paget’s disease predispose and LR6 region which was subsequently to osteomyelitis. Systemic diseases like drained. The pain remained poorly diabetes, anaemia and malnutrition controlled despite liberal quantities of that cause concomitant alteration in Oramorph and MST. As time progressed host defences profoundly infl uence the 10 Fig. 5 One year later. Axial CBCT slice the infection began to tract further for- course of osteomyelitis. The incidence through the level of the mandible. Showing ward and pus was evident in the gingivae of the disease has decreased dramatically good repair of the left lingual cortical plate, of the anterior teeth. The patient sought a with the introduction of antibiotics and however, a buccal area of perforation is now second surgical opinion and at this time improvement in the general health to the more pronounced (arrowed)

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population together with access to medical Differential diagnosis The outstanding clinical character- and dental care.11-13 The differential diagnoses of yet to istics of the two cases were the intense The jaws are unique from other diagnose PCO includes malignant and and uncontrollable nature of the pain bones of the body in that the pres- benign entities discussed by Eyrich et with little or no accompanying physi- ence of teeth creates a direct pathway al.,6 Baltensperger et al.22 and Soubrier cal signs. Infl ammatory indicators were for infectious and infl ammatory agents et al.23 The benign include ossifying and normal. The disparity between signs and to invade bone by means of caries and non-ossifying fi broma, infection of the symptoms were so great as to make the periodontal disease.14 Oral bone appears salivary glands (juvenile recurrent paro- clinician doubt the veracity of the patient’s to be particularly resistant to infection titis or chronic recurrent sialadenitis) and history. The combination of MRI and CBCT despite exposure to oral fl ora.15 This fur- non-specifi c chronic lymphadenitis. The examination were helpful in distinguish- ther reiterates the rarity of the mandible malignant entities that should be consid- ing changes in the bone. The lesson drawn experiencing osteomyelitis. ered because of the insidious nature of PCO from these cases is that in the early stages are Ewing’s sarcoma, osteosarcoma, chon- of chronic osteomyelitis, the identifi cation Microbiology drosarcoma, non-Hodgkin’s lymphoma of the disease depends largely on clini- Osteomyelitis of long bones is normally and metastatic disease. cal judgement rather than haematological attributed to Staphylococcus aureus and radiographic tests. Another character- whereas in mandibular osteomyelitis it Pathogenesis istic was the reluctance of the infection is usually considered a polymicrobial The varied treatments for PCO refl ect the to respond to standard regimen of oral disease.8 The search for an infectious lack of understanding of the aetiology antibiotics possibly due to the pathogen- aetiological agent of PCO has led some of this disease. It is thought the rela- esis theory proposed earlier. Rather long researchers to investigate the microbio- tively avascular and ischaemic nature of courses of IV antibiotics are required to logic samples taken from surgical speci- the infected region and sequestrum pro- resolve the infection. Oral antibiotics mens. Bacteriologic and serologic studies duces an area of lowered oxygen ten- seem ineffective. have shown Propionibacterium acnes,16 sion as well as an area that antibiotics The role of an intra-osseous injection Actinomyces species, or Eikenella corro- cannot penetrate. The lowered oxygen in the induction of osteomyelitis remains dens17 as causative agents, but cultures tension effectively reduces the bacte- unclear. Published literature has stated from the bone lesions often show negative riocidal activities of polymorpholeuko- symptoms of pain and swelling post results18,19 and no specifi c microorganism cytes and also favours the conversion administration of a intra-osseous injection has been identifi ed as a dominant aetio- of a previously aerobic infection to one post-operatively.28-30 Furthermore Replogle logical agent.11-13 This therefore shows the that is anaerobic. The diffusion rate of et al.29 reported purulence following intra- differential between osteomyelitis in long antibiotics into dead bone is so low that osseous injection which resolved up to 14 bones and the mandible. Where in long frequently it is impossible to reach the days post administration without any mor- bones infection is via Staphylococcus organisms regardless of the external con- bidity. This form of analgesia has not been aureus which is usually transferred via centration. This may lead to ineffective associated with osteomyelitis in the medi- the bloodstream, this has proven not to be antibiotic concentrations at the site of cal literature. However, it was obvious as the case when the mandible is affected. infection despite serum levels indicating the instigating factor in the second case. therapeutic concentrations.24 It remains a mystery why a healthy adult Imaging patient should develop osteomyelitis after There remains much choice when con- Treatment a simple intra-oral injection. sidering imaging for osteomyelitis. A Treatment varies from a range of simple simple dental panoramic radiograph may non-invasive approaches to more inva- CONCLUSION be enough to diagnose this condition. sive and radical treatment. The nonsur- Osteomyelitis remains a rare entity in med- However, the disease process may only gical approach includes: antibiotics,23 ically fi t and well individuals. The clinical become evident on the radiograph in the NSAIDS,23 hyperbaric oxygen therapy,25 features in these patients are not typical latter stages. MRI T1 weighted images bisphosphonate treatment,15,23 and muscle of those seen in the traditional debilitated are usually better as infl amed tissue cre- relaxants.18 Following the failure of a non- patient and can pose a diagnostic problem. ates low signal intensity in the normally surgical approach a surgical intervention Osteomyelitis should always be considered bright signal of fat contained in the mar- to consider include decortications alone,25 in the presence of intense and poorly con- row.2 MRI does not show specifi c features decortication with bone grafting,26 par- trolled pain following injury to the jaw. capable of making a diagnosis, but does tial (marginal) resection,27 and segmental Clinicians should remember that osteomy- show the extent of the lesions and may resection.23,27 Unfortunately, conservative elitis responds poorly to antibiotics and be helpful in disease monitoring.20,21 The management invariably could lead to may require long term IV and oral doses, use of cone beam CT enables an image multiple recurrences of the disease, and possibly even as multiple courses. Finally, of high quality of a selected area. This aggressive management may lead to sig- consideration of CBCT as part of radio- imaging was used for the cases described nifi cant co-morbidity with subsequent logical examination may help conclude a above and proved to give accurate and need for reconstructive surgery7 therefore diagnosis earlier due to the localisation of detailed information. leaving the clinician with a dilemma. the imaging.

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