1J Med Adv Clin Case Rephttps://www.jmaccr.com

CASE REPORT

Received: 06 April 2020 Accepted: 28 April 2020 Published: 04 May 2020

Maxillary and Mandibular Osteomyelitis, Two Unexpected Cases in Healthy Patients

Mauricio Barreda1* | Javier Cuéllar1 | Daniel Pino2 | Pablo Romero2

1Oral and Maxillofacial Surgery Service, Hospital Clínico San Borja Arriarán, Chile

2Oral and Maxillofacial Surgery Service, Hospital del Salvador, Chile

1 INTRODUCTION

Osteomyelitis is an inflammation of bone tissue which begins with an infection of the central component that engages the Haversian system, quickly spreading to the periosteum [1]. Odontogenic infections can spread by continuity causing os-teomyelitis of the jaws. Anatomical conditions of the mandible make it more susceptible than the maxilla due to less vascular-ization, reduced bone marrow and cortical thickness [2]. How-ever, the occurrence of this type of infection is low. Cases in which it usually occurs are related to a predisposing condition such as fracture, radiation, immunosuppressive states; HIV, malnutrition, diabetes mellitus corticosteroids, making even more infrequent the occurrence of osteomyelitis in a healthy patient [3]. The most frequent causes of maxillary osteomyeli-tis are dental infections and sinusitis, followed by trauma and radiation [2].According to time we can distinguish an acute and a chronic entity. Osteomyelitis develops within one month and is con-sidered chronic when extended beyond this period [4]. Once the infectious process is established in the bone, intramedul-lary pressure increases, generating a drop in the blood supply, which worsens the clinical scenario by decreasing the immune response, added to tissue ischemia that culminates in a bone necrosis. The formation of purulent material accumulated un-der the periosteum may eventually perforate this barrier de-veloping abscesses and mucous or cutaneous fistula. If the

process becomes chronic, non-viable tissue is isolated forming sequestrum. Pain is a common symptom and also anesthesia or hypoesthesia may be present [5].In maxillofacial osteomyelitis, microbiology involves patho-gens also present in odontogenic infections, such as Fusobac-terium nucleatum in suppurative processes, Prevotella, Pep-toestreptoccous, Streptococcus and Actinomyces sp. Except for patients with comorbidities Gram negative bacilli, such as Staphylococcus aureus, are uncommon.Treatment is usually complicated because of the presence of teeth and a constant exposure to a contaminated medium, the oral cavity. Surgery with prolonged antibiotic therapy, weeks or months, is required. Surgical treatment is usually debride-ment, removal of bone sequestrum, surgical scrubbing and pri-mary closure [4,5].

2 CASE REPORTS2.1 CASE 1

A 28-year-old man, presented to the Maxillofacial service complaining of discomfort and bad smell in left maxillary with a year of evolution, without significant morbid history. He re-ferred having an odontogenic infection two years before con-sulting and endodontic treatment was performed on tooth 26. One year after; endodontic treatment couldn’t solve the infec-tion, so tooth extraction was performed. There was increased

Journal of Medical & Advanced Clinical Case Reports

CorrespondenceDr. Mauricio Barreda, DDSOral and Maxillofacial Surgery ServiceHospital Clínico San Borja ArriaránChileE-mail: mbarredah@gmail.com

AbstractBackground: Osteomyelitis corresponds to an inflammation of bone tissue, which begins with bone marrow infection. Generally, it affects the mandible and develops under circumstances that favor the appearance of this condition, such as radiation, immunosuppressive states, diabetes, HIV, malnutrition and corti-costeroid therapy. It can also occur after mandible fractures and their treatment. The manifestation in the healthy population is a rare entity. Microbial etiology is usually pyogenic germs, such as Staphylococci and Streptococci and in some occasions, Pneumococci and Enterobacteria. Actinomyces is not a common flora that generates osteomyelitis.Case Presentation: We report two unusual cases of maxillofacial osteomyelitis in healthy patients, where the causative agent isolated was Actinomyces.Conclusion: Osteomyelitis in the maxillofacial area is an uncommon manifesta-tion. Actinomyces could act as pathogen protagonist or appears secondarily to a preexisting osteomyelitis generating a favorable environment for pathogens growth.KEYWORDSMaxillary, Mandibular, Osteomyelitis, Healthy patient

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and ceftriaxone 2g EV, removal of necrotic tissue and in-volved teeth 24, 25 and 27. Closure was done by transposition of the pediculate buccal fat pad and L-PRF. Histopathologic-al study of the removed specimen reported mature trabecular bone tissue with Actinomyces colonies in medullary spaces. In addition, inflammatory tissue suggesting a chronic sinus-itis and areas of fibrosis. Gram and Hematoxylin and eosin stain confirmed the presence of Actinomyces (Figure 3). The patient was discharged from hospital with antibiotic treatment of amoxicillin 1g for 1 month. Control at five days, two weeks, three weeks, two months and six months reflect a favorable evolution (Figure 4).

2.2 CASE 2

A 52-year-old woman was sent from the Emergency Depart-ment due to a mandibular infectious event. She presented no significant medical history or use of medications or drugs. The patient reported endodontic treatment of tooth 46 sub-sequently continuing with pain, so antibiotics and analgesia were prescribed. The symptoms persisted for 5 days. She was then hospitalized for intravenous ATB administration and was discharged after 5 days. However, infection persisted and the patient was referred to the Maxillofacial Surgery Service. The presence of active fistula in the right molar region was clinic-ally established (Figure 5) and the radiographic examination revealed the presence of a diffuse radiolucent area. CT scan showed the presence of bone sequestrum (Figure 6). The treat-ment plan was administration of intraoperative antibiotics, clindamycin 600 mg IV plus ceftriaxone 2g IV, removal of necrotic tissue and teeth 46 and 47. The wound was treated by primary closure. Histopathological study of the removed speci-men showed necrotic bone Actinomyces forming hyphae-like structures (Figure 7). The patient had satisfactory evolution,

volume compatible with abscess, increased pain symptoms and fever. Unfortunately, the infection, persisted and pro-gressed. Clinical examination showed that bone sequestration occurred between tooth 24 and 27, with extension to the lateral wall of the maxillary sinus (Figure 1). Blood count, biochem-ical profile and coagulation tests were within normal param-eters. PCR and ELISA tests for HIV were negative. CT scan showed maxillary bone involvement from teeth 24 to 27, with loss of buccal cortical bone and some hypodense content in the left maxillary sinus (Figure 2). The treatment plan was intrao-perative antibiotic administration of clindamycin 600 mg EV

Figure 1: Necrotic bone and sequestrum extending from tooth 24 to 27

Figure 2: A) Coronal view showing the left maxillary sinus affected; B) Postoperative image, necrotic bone was removed

Figure 3: A) Gram stain confirms the presence of Actinomyces; B) Hematoxylin and eosin stain also confirms presence of Actinomyces

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being discharged under antibiotic treatment for 3 months.Radiographic controls after 2 years revealed diffuse lesions in teeth 45, 44 and 43, compatible with aseptic tooth necrosis and the patient was referred for endodontic treatment of those teeth. (Figure 8). Currently, she presents no lesions.

3 DISCUSSION

Presently, osteomyelitis of the maxillofacial region corres-ponds to a relatively rare infection of bone tissue, especially since the introduction of antibiotics [1]. The literature reports

many cases with several clinical presentations, where the com-mon pattern is an inflammation and infection of bone, under systemic conditions which favors it development. Specially immune suppressive situations, such as HIV, uncompensated diabetes mellitus, malnutrition, anemia and factors that could compromise the vascularization of the bone, such as radiation, neoplasms, osteoporosis, osteopetrosis and Paget’s disease, have been implicated in its etiology [6].Usually the occurrence is higher in the mandible than in the maxilla. However, Peravali, in a report of 31 cases, found greater maxillary than mandibular involvement (1.07:1) and 70.9% of cases (22 patients) had a predisposing medical con-dition (diabetes, anemia and malnutrition).The literature describes several types of classifications [6]. We considered those focused on an acute, subacute and chronic phase [3]. An abrupt onset of signs and symptoms during the initial stages indicate acute osteomyelitis. If during this phase, untreated or inadequately treated, a complete elimination of infection does not occur, a subacute or chronic osteomyelitis stage is established. Chronicity of osteomyelitis is multifactor-ial. Vascular conditions and onset of ischemia in the infected site determine the appearance of sequestrum, promoted by lack of vascularization and low oxygen supply. Penetration and antimicrobial activity of leukocytes decreases and the for-mation of an anaerobic environment occurs [6].Conventionally, the etiology of osteomyelitis has been associ-ated with bacterial infections. But it could also be the result of a fungal, parasitic or viral presence. Staphylococcus aureus is the most frequent pathogen, that could come from the skin and colonize deeper tissues secondarily to surgery or trauma. The spread of the infectious process can occur by contiguity or by hematogenous route [6].Infections from maxillofacial origins usually present a polym-icrobial mixed flora, both aerobic and anaerobic [1]. It includes pathogens such as Streptococcus, Bacteroides, Peptostrepto-coccus and other opportunistic pathogens. However, Eikenella and Actinomyces have been unusually described, presented as contaminating pathogens of the original infection [7]. The most common route of spread is usually hematogenous, but head and neck osteomyelitis differ from the rest of the body by the presence of sinuses and teeth. Those represent a direct path for bacterial invasion from the necrotic pulp, periodontal tissues and respiratory mucosa, facilitating contiguous spread to the bone [3]. In fact, any procedure in oral surgery, pulpal necrosis periodontal disease or facial trauma, among others, can participate as inoculation for the entry of microorganisms.In the cases of chronic osteomyelitis of dental origin pre-sented, Actinomyces was identified as the causative agent. The origin of Actinomycosis in osteomyelitis is not clear, it is suggested that an inflammation begins when the normal

Figure 4: Postoperative image after six months, showing healthy wound and asymptomatic patient

Figure 5: Intraoperative image showing active fistula

Figure 6: A) Coronal section showing necrotic bone and sequestrum on right mandibular body; B) Axial section

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composition of the microbial flora is disturbed and chronicity leads to pathological changes located in the bone, which pro-motes their growth [8]. Actinomyces is a facultative anaerobic gram-positive Bacillus associated with infectious processes of the CNS, lungs, chest and abdomen. It also corresponds to a regular presence in the flora of periodontal pockets, cavities and tonsillar crypts. Secondarily to an injury or gap in the mucosa, it can invade surrounding structures of the head and neck regions, as a result of dental infections and maxillofacial trauma [5].Overall, actinomycosis is a rare disease that can occur in any age and is most frequent between 30 and 60 years of age. For unknown reasons, it affects more men than women with a 4:1 ratio [9]. The most common form of infection is cervical fa-cial actinomycosis (50% of cases), followed by abdominal cavity (20%) and thoracic cavity infections (15%) [5]. Those are not opportunistic infections, but their occurrence is asso-ciated with predisposing immune suppressant conditions sim-ilar to those of osteomyelitis [10]. Bahar Sezer, mentions that Actinomyces has a minimal invasive potential due to absence of hyaluronidase and low virulence, thus requiring disruption of the oral mucosa [11] and a polymicrobial ecosystem of ac-companying bacteria, as well as synergistic pathogenicity, to create a clinically infection [9]. This microflora works together

destroying vascularized tissue aerobically and creating a poor oxygen granulation tissue. In the jaw bone Actinomyces in-fection is usually secondary to a preexisting process. Similar to our report (Case 1), Hirshberg mentions that a periapical lesion that is unresolved after endodontic treatment could pro-vide reservoirs of Actinomyces [10,12] and describes a case of maxillary osteomyelitis by Actinomyces where the inoculation was through a tooth root after pulpal necrosis.During the development of osteomyelitis, pus is gradually formed and propagates to vascular channels. Affecting intraos-seous pressure, increasing blood flow. This process will result in an intense inflammatory response, purulent thrombosis and the subsequent dissection in the periosteum, leading to sub-periosteal abscesses and non-vascularized bone fragments [13].Microorganisms, neutrophil infiltration and congested or thrombosed blood vessels are the main histological findings in acute osteomyelitis [13]. Once a balance between bone resorption and new bone formation is achieved, the chronic phase osteomyelitis occurs [14]. Clinically, the acute phase is characterized by the presence of a fluctuating mass, volume increase and often mimics odontogenic infection, with quick spread through tissues. Signs and symptoms, such as fever and pain are usually present. On the other hand, the chronic form, most common, is characterized by slow progression and development of an inflammatory mass. It may or may not be painful. Laboratory tests could result normal or slightly altered [9].The definitive diagnosis is based on clinical, radiologic, and histopathologic findings. Bacteriological cultures for Actino-myces are often difficult to obtain, due to their demanding con-ditions of anaerobic growth and overgrowth of other bacteria [15]. The presence of Actinomyces does not confirm a lead etiological role, as this may be just late contamination [12]. So, it is difficult to decide whether Actinomyces was primari-ly responsible for the infection or a late colonizer of necrotic bone [10].Treatment is usually complicated by the presence of teeth and constant exposure to a contaminated medium such as the mouth. It should be considered surgical debridement of necrot-ic bone, combined with prolonged antibiotic therapy for weeks or months. Antimicrobial drugs used are usually penicillin and derivates, tetracyclines, clindamycin or cephalosporins. Ac-tinomyces usually show sensitivity to penicillin and erythro-mycin [16]. 4 CONCLUSION

In the maxillofacial region osteomyelitis is an uncommon occurrence. The incidence on the maxilla or mandible bear relationship with anatomical and vascular characteristics of those bones. Systemic predisposing conditions may favor the development of such infections. However, they may occur in healthy patients. The flora involved is similar to odontogenic polymicrobial infections. Actinomyces could act as a protag-onist pathogen or appear secondarily in a preexisting osteo-myelitis.

5 REFERENCES

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Figure 7: Histopathological study, HE: Indicates necrotic bone and hypha-like structures of Actinomyces

Figure 8: A) Two years postoperatively. Periapical lesions (white arrow); B) Four years postoperative view

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