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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 9  |  Issue : 3  |  Page : 205-209

Mandibular actinomyces osteomyelitis


1 Division of Maxillofacial Surgery, University Multiprofile Hospital for Active Treatment and Emergency Medicine “N. I. Pirogov”, Sofia, Bulgaria
2 Department of Anatomy, Histology and Embryology, Medical University of Sofia, Bulgaria
3 Department of General and Clinical Pathology, Medical University of Sofia, Bulgaria

Date of Submission14-Nov-2022
Date of Decision12-Dec-2022
Date of Acceptance13-Dec-2022
Date of Web Publication31-Dec-2022

Correspondence Address:
Dr. Bistra Blagova
Division of Maxillofacial Surgery, University Multiprofile Hospital for Active Treatment and Emergency Medicine “N. I. Pirogov,” Gen. Totleben Blvd. 21, 1606 Sofia
Bulgaria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/sjoralsci.sjoralsci_50_22

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  Abstract 


Actinomycosis caused by the Actinomyces genus can affect both soft and bone tissues. Its diagnosis depends on positive culture or identification of Actinomyces colonies and sulfur granules in histological specimens. This article aims to report a clinical case of actinomycosis in the mandible. The diagnosis of actinomycosis osteomyelitis was confirmed by bone biopsy in a female with a long-standing history of generalized periodontal disease. She underwent debridement of necrotic tissue in the region, and the material was sent for histopathological analysis, which revealed chronic localized actinomycosis osteomyelitis. The patient responded well to prompt systemic antibiotics and local surgical measures with complete resolution of the infection. Clinicians should be aware of the possibility of actinomycosis arising in generalized periodontal disease and the importance of bone biopsies and cultures in arriving at a definitive and timely diagnosis. The accurate diagnosis of actinomycosis is crucial for the successful treatment outcome.

Keywords: Actinomycosis, alveolar bone loss, osteomyelitis, periodontal disease


How to cite this article:
Blagova B, Malinova L, Ivanova V. Mandibular actinomyces osteomyelitis. Saudi J Oral Sci 2022;9:205-9

How to cite this URL:
Blagova B, Malinova L, Ivanova V. Mandibular actinomyces osteomyelitis. Saudi J Oral Sci [serial online] 2022 [cited 2023 Feb 6];9:205-9. Available from: https://www.saudijos.org/text.asp?2022/9/3/205/366535




  Introduction Top


Actinomyces genus is filamentous Gram-positive microaerophilic (anaerobic or facultatively anaerobic) saprophytic bacilli that are commensal organisms of the oropharynx, gastrointestinal tract, and urogenital tract.[1] These organisms cause odontogenic and cervicofacial infections, including gingivitis, periapical abscesses, periodontitis, and surrounding soft-tissue infection. Still, they are occasionally the causative organism in pneumonia, empyema, abscesses, osteomyelitis, and pericarditis.[2]

Osteomyelitis of the mandible is a rare complication of odontogenic infections that frequently develops from the contiguous spread of chronic dental disease with Actinomyces.[2] Diagnosis is particularly challenging, as Actinomyces osteomyelitis closely resembles other infectious and noninfectious etiologies, both clinically and radiographically.[2] For this reason, our article aims to present a case of mandibular osteomyelitis combined with generalized periodontal disease caused by Actinomyces with an unclear initial route of infection and predisposing factors.


  Case Report Top


A 57-year-old female sought treatment with a primary complaint of a “loose tooth” sensation in her lower jaw. The patient was immunocompetent, and her medical history was noncontributory. She denied fevers, chills, night sweats, or other constitutional symptoms. Her dental history was significant for generalized periodontal disease with teeth loss. Her head and neck lymph nodes were within the normal limits on physical examination. After removing the patient's poorly-fitting mandibular denture, the intraoral evaluation revealed bone expansion buccally and lingually in a severely luxated lower right canine area. There was mild purulent discharge from the surrounding gingival sulcus. The overlying soft tissue was without ulcerations. There were no fistulizing tracts. The patient's panoramic radiograph revealed defined bony changes with osteolytic and osteosclerotic areas, i.e., bony sequestrum within a radiolucent capsule in the right mandibular premolar region [Figure 1].
Figure 1: Preoperative panoramic view showing radiolucent and radiopaque areas in the right mandibular premolar region

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A full blood profile, including erythrocyte sedimentation rate and C-reactive protein, were reported as normal. Because of concern for chronic infection, the patient underwent debridement under general anesthesia. Purulence and necrotic bone were observed during this procedure. Curetted bone tissue and the canine from the surgical site were submitted for histopathological and microbiological examination. The operative field was repaired with a simple advancement flap. Postoperatively, empirically the patient was treated with oral amoxicillin combined with clavulanic acid 875 mg twice daily and a chlorhexidine gluconate oral rinse for 2 weeks. No postoperative complications occurred. The patient was discharged on the 2nd postoperative day. She responded well to antimicrobial treatment with complete healing of the operative site without any signs of infectious relapse. Based on histological results, antibiotics continued for 4 weeks.[3] The patient was referred to her dentist for dental treatment and mouth rehabilitation. Currently, this patient is symptom-free and under long-term review.

The microbiological culture of the tissue was positive only for Streptococcus alpha heamoliticus. However, the histopathological examination of the biopsied bone revealed granulation tissue with abundant Gram-positive filamentous organisms suggestive of actinomycosis. In H and E (H and E) stained, confirmed by Periodic acid–Schiff (PAS)-staining, bone, and granulation tissue was observed. Hard-tissue specimens included trabeculae of bone enclosing marrow tissue with extensive involvement of Actinomyces granules; nonspecific inflammatory cell infiltrates embedded in an abscess; vascular proliferations and granulation tissue were seen. The periphery of the Actinomyces granules showed radiating, basophilic filaments and eosinophilic, club-shaped ends. Within the granulation tissue were granules surrounded by polymorphonuclear leukocytes. Actinomyces granules were observed with bone marrow and granulation tissue [Figure 2].
Figure 2: Histopathological features consistent with infection by actinomyces organisms: A photomicrograph demonstrating granulation tissue with multiple sulfur granules: (a) Hematoxylin-Eosin (H-E), and (b) Periodic acid-Schiff (PAS), ×20

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  Discussion Top


Jaws are unique from other body bones in that the presence of teeth creates a direct pathway for infectious and inflammatory agents to invade bone using caries and periodontal disease.[4] Osteomyelitis can be defined as an inflammatory condition of the bone, which begins as an infection of the medullary cavity, rapidly involves the Haversian systems, and extends to the periosteum of the affected area.[5] Osteomyelitis of the jaw is a relatively uncommon inflammatory disease in developed countries.[6] The etiology is unknown, and theories include bacterial infection (dental or bacteremia from distant foci), vascular deficiency (localized endarteritis), autoimmune disease, or trauma.[7],[8] The search for an infectious etiological agent of primary chronic osteomyelitis has led some researchers to investigate the microbiologic samples taken from surgical specimens. Bacteriologic and serologic studies have shown Propionibacterium acnes, Actinomyces species, or Eikenella corrodens as causative agents, but cultures from the bone lesions often show negative results, and no specific microorganism has been identified as a dominant etiological agent.[9],[10],[11],[12]

Historical notes

Historically, von Langenback may have been the first researcher to describe the first incidence of Human actinomycosis in 1848, and he attributed it to a fungus. In 1891, Israel and Wolf isolated an anaerobic filamentous organism in humans. In 1898, this microorganism received the name Actinomyces israelii. In 1960, Waksman demonstrated that Actinomyces were Gram-positive bacteria.[13] Bollinger described the organism Actinomyces bovis and its ability to cause a “lumpy jaw” in cattle. Actinomycosis is a Greek word comprising “aktino” meaning radiating appearance of sulfur granules, and “Mykonos” which labels the condition as a mycotic disease. Hence, the word Actinomyces means “ray fungus” and reflects the general belief at the time that the organism was a fungus. Actinomyces strains resemble both bacteria and fungi. Thus, they were often considered transitional between the two groups of microorganisms. However, most of the fundamental characteristics of Actinomyces indicate that they are, in fact, bacteria.[14]

Actinomycosis infection can be divided into three categories: cervicofacial (55%), abdominopelvic (20%), and thoracic (15%), depending on the region.[15] Cervicofacial infections are the most common manifestation of actinomycosis, although this is generally limited to the soft tissues without spreading to involve neighboring bone.[1] Only a few cases of actinomyces osteomyelitis have been reported in the literature.[16] Primary actinomycotic osteomyelitis is rare, corresponding to about 12% of cases.[1] The prevalence ratio in the mandible to the maxilla is 4:1.[16]

Etiopathogenesis

The principal cause of cervicofacial actinomycosis is A. israelii. However, Actinomyces naeslundii, Actinomyces viscosus, and Actinomyces odontolyticus are occasionally identified.[16] The exact pathogenic mechanism underlying actinomycotic bone infiltration is unknown.[13] Actinomyces produces chronic, slowly developing infections, particularly when standard mucosal barriers are disrupted by trauma, surgery, or primary infection, and the presence of devitalized tissue can result in an invasion of the deeper body structures and cause illness.[14] Actinomycosis is generally a polymicrobial infection requiring the presence of companion bacteria, most frequently anaerobic Streptococci, fusiform or Gram-negative bacilli, and Haemophilus species, as it was confirmed in the presented case as well.[17],[18] The associated flora form a kind of symbiosis with Actinomyces species and may cause an anaerobic environment which furthers the growth of this species.[18] Hence, these associated bacterial species act as co-pathogens and participate in the production of infection by elaborating a toxin or enzyme or by inhibiting host defenses.[19] Furthermore, these accompanying species enhance the relatively low invasive power of Actinomyces by eliciting early manifestations of the infection.[20]

Predisposing factors

Predisposing factors are local and systemic. The most common inciting events are dental caries, dental manipulation, maxillofacial trauma, or pressure ulceration caused by poorly fitting prosthetics facilitating mucosal breakdown and spread of infection.[21] Other risk factors for acquiring Actinomyces infections include poor oral hygiene, diabetes mellitus, steroid and bisphosphonate therapy, leukemia with chemotherapy, human immunodeficiency virus coinfection, prior transplant with immunosuppression, and heavy alcohol intake.[22],[23] Our patient, in her turn, was immunocompetent, and her clinical history was not contributory, resembling some reports found in the literature.[24] Otherwise, she revealed a generalized periodontal disease with a periodontal pocket that could be the portal for Actinomyce's access into deeper bone tissue.[25]

Clinical manifestation

The clinical presentation may be either acute or chronic. The acute infection occurs less frequently and may exhibit floating swelling, pain, and fever. On the other hand, a chronic condition is more common and may evince a slow progressive volume increase, with or without symptoms.[23],[26] Unlike most cervicofacial infections, regional lymphadenopathy is uncommon, as observed in the presented case.[19] However, any unidentified mass, facial swelling, or persistent infection, particularly after endodontic therapy or tooth extraction, should suggest actinomycosis, regardless of its nontraumatic history.[27]

Differential diagnosis

The diagnosis is particularly challenging as actinomyces osteomyelitis mimics several infectious and noninfectious pathologies.[2] The differential diagnosis includes osteosarcoma, metastases, lymphoma, and diseases involving granuloma formation, such as Langerhans cell histiocytosis, sarcoidosis, tuberculosis, and fungal infections.[2],[24]

Diagnostic criteria

Although history and physical examination are essential to diagnosing any disease, in the case of actinomyces osteomyelitis, other diagnostic methods are used, such as image examinations and examination of bacterial culture and cytopathologic assessment of tissues and secretions collected from the infection site.[16],[26]

Radiographs can be helpful in the infectious process extension recognition in bone but are nonspecific for actinomycosis.[26] A simple dental panoramic radiograph may be enough to diagnose this condition.[28] However, it is challenging to differentiate mandibular osteomyelitis from other bone lesions.[2],[24]

A diagnosis of actinomycosis is best made by culture, but <50% of cases are favorable due to numerous problems associated with culturing these organisms.[29] For this reason, a histopathological examination is also highly recommended.[30] Herein, the diagnosis depends on the morphology and staining characteristics of the microorganism.

Bone biopsy is the most specific method for determining the nature of actinomyces osteomyelitis.[31] Actinomyces species strongly stain positive for H and E stain, PAS, and Giemsa.[32] Still, the outcome of the examinations is only sometimes constant.[25] Histopathological findings suggestive of actinomyces infection are Gram-positive filamentous rods, which are collections of inflammatory cells surrounded by hyphae, degenerated bacteria, and proliferating fibroblasts. There are peripheral club-shaped, sometimes eosinophilic, structures compatible with sulfur granules characteristic of infection by Actinomyces spp.[22],[26] It is important to note that sulfur granules are commonly missed.[33]

Management

Early diagnosis and vigorous broad-spectrum antibiotic treatment with surgical debridement is the primary form of therapy.[25] Stand-alone conservative management is not always curative. After arriving at a proper diagnosis, it is recommended that treatment of actinomycosis infection should be vigorous by removing the foci of infection, including resection of the sequestrated bone and curettage of all granulation tissue until healthy tissue is exposed. This is required for removing necrotic tissue and penetration of antibiotics into the colony of microorganisms, which is inaccessible otherwise, either because of fibrous tissue or surrounding edematous tissue. Historically, surgical debridement has played a role in areas of necrotic tissue, fistulizing sinus tracts, and cases, in whom malignancy is difficult to exclude, such as in our case.[23] After removing the foci of infection, additional exposure time to antibiotics is necessary because lysis of Actinomyces species occurs slowly compared to most other bacteria.[3],[30],[34] Treatment regimens should be individualized based on individual clinical presentation.[34] Factors that affect the duration of treatment include the initial burden of disease, the site of infection, the extent of surgical debridement, and the clinical and radiological response to therapy.[2] Close follow-up is essential in determining the trajectory of the treatment course and the need for surgical revision.[2],[25] Regular visits to the dentist are fundamental to evaluating possible recurrences since actinomycosis can relapse years after contamination by Actinomyces or years after the inadequate first treatment.[22]


  Conclusion Top


The presented case demonstrates an association between two chronic, destructive, and disfiguring conditions of the mandible, namely generalized chronic periodontitis and Actinomyces osteomyelitis. However, it was not clear whether the actinomycosis was the primary infection or a secondary infection to a preexistent nonspecific local infection of the alveolar bones. Actinomyces osteomyelitis of the mandible could be an unusual sequel of odontogenic infections. The diagnosis often is overlooked because of its ability to mimic other conditions, and its presentation often overlaps with other infectious and noninfectious disease both clinicoradiologically. The clinical features in these patients are not typical of those seen in the traditional debilitated patient and can pose a diagnostic problem. Clinicians should be aware of the possibility of actinomyces osteomyelitis and the importance of histopathology in arriving at a definitive and timely diagnosis. When actinomycosis is clinically suspected, a biopsy is recommended because some cases cannot be diagnosed by typical microbiological culture tests.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published and due efforts will be made to conceal identity but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
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Russo TA. Agents of actinomycosis. In: Mandell, Douglas, and Bennett's Principles and Practice of Infectious Diseases. Updated ed. Churchill Livingstone Elsevier Inc, Philadelphia; 2015. p. 2562864-73.  Back to cited text no. 33
    
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