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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 11  |  Issue : 3  |  Page : 151-155

Actinomycotic osteomyelitis of mandible: An infrequent case presentation


Department of Oral Pathology and Microbiology, King George's Medical University, Lucknow, Uttar Pradesh, India

Date of Submission03-Jun-2020
Date of Acceptance20-Aug-2020
Date of Web Publication15-Oct-2020

Correspondence Address:
Dr. Shruti Singh
Department of Oral Pathology and Microbiology, King George's Medical University, Lucknow, Uttar Pradesh
India
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DOI: 10.4103/srmjrds.srmjrds_47_20

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  Abstract 

Actinomycosis is a rare entity causing acute or chronic infections by filamentous bacteria, Actinomyces species. It usually affects soft tissue but rarely affects bone leading to Actinomycotic osteomyelitis. We report a case of 22-year-old male clinically and radiologically diagnosed as Osteomyelitis, and histopathologically confirmed as Actinomycotic osteomyelitis. The current study also highlights the importance of histopathological examination of any clinically suspected lesion, due to low biological identification rate. Correct diagnosis of the disease avails adequate treatment and thus better prognosis.

Keywords: Actinomycosis, cervicofacial, head and neck, jaws, mandible, osteomyelitis


How to cite this article:
Singh S, Singh J, Chandra S, Fatima T, Mehta MK, Gupta N. Actinomycotic osteomyelitis of mandible: An infrequent case presentation. SRM J Res Dent Sci 2020;11:151-5

How to cite this URL:
Singh S, Singh J, Chandra S, Fatima T, Mehta MK, Gupta N. Actinomycotic osteomyelitis of mandible: An infrequent case presentation. SRM J Res Dent Sci [serial online] 2020 [cited 2020 Oct 25];11:151-5. Available from: https://www.srmjrds.in/text.asp?2020/11/3/151/298264


  Introduction Top


Actinomycosis is a rare, subacute or chronic infection caused by filamentous gram-positive, microaerophilic bacilli and a common commensal of oral flora, the Actinomyces species.[1] Actinomycosis usually characterized by a chronic disease resulting in multiple abscesses, firm soft-tissue masses, and the presence of sulfur granules in exudates or tissues.[2] It mainly affects the soft tissues and bone involvements are a rare finding.[3] Within bone, it manifests as osteomyelitis.[3] Actinomycotic osteomyelitis accounts for about 12% of total osteomyelitis.[4] Actinomycosis can be divided in three categories: cervicofacial (55%), abdominopelvic (20%), thoracic (15%) depending on the region. Cervicofacial actinomycosis is the most common type.[5] The principle cause of cervicofacial actinomycosis is Actinomyces israelii. However, Actinomyces naeslundii, Actinomyces viscosus, and Actinomyces odontolyticus are occasionally identified.[6]

Actinomycosis usually presents as an indolent course with painful or painless swelling of the surrounding soft tissue that may or may not be painful. Diagnosis of cervicofacial Actinomycosis is perplexing due to its resemblance with other etiologies in its clinical and radiological behavior and also due to its rarity.[7]

The current case report illustrates a diagnostically challenging case of mandibular Actinomycotic osteomyelitis. Since the prevalence of actinomycotic osteomyelitis of the jaw is scarcely reported in the literature and also, in this exclusive case, only a small section of the biopsied specimen showed colonization of the microorganisms, this emphasizes the need of careful histopathological examination in order to achieve accurate diagnosis and thus the correct treatment protocol.


  Case Report Top


A 22-year-old male patient walks into the outpatient unit of our institution with the chief complaint of swelling in the right side of the lower region of the face. The onset was slow and gradual progressive lesion and the total duration of the lesion was 7 years. Along with the swelling, other symptoms included pain with fever and subject was unable to hear from the right ear. The patient's family history was noncontributory while the patient had a medical history of epilepsy and was under medication for the same for 3 years.

Extraoral examination revealed facial asymmetry due to swelling along with multiple sinus openings with scarring of the sinus openings. The swelling anteroposteriorly extends from the corner of the mouth through the auricle till the mastoid region. Superiorly, it extends from the inner canthus of the eye and inferiorly extends beyond the lower border of the mandible involving the neck region. Lymph nodes were palpable, tender, mobile, and hard in nature. Intraoral findings showed caries in 48 [Figure 1] and [Figure 2].
Figure 1: Clinical Picture of the patient showing facial asymmetry on left side; the swelling extending from outer canthus of the eye till submandibular region (front view)

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Figure 2: Clinical picture of the subject showing unilateral multiple scarred sinus openings from corner of the mouth crossing the mastoid region till the cervical area. (side view)

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Computerized tomography revealed evidence of expansile osteo-sclerotic lesion with ground glass opacification involving right mandible, pterygoid plates, right temporal bone, basi-sphenoid, and zygomatic bone. Large ill-defined, soft-tissue lesion with enhancing areas in the right side of the neck extending from submandibular region to right side lower cervical region along with right-sided otitis media and mastoiditis. Magnetic resonance imaging (imaging parameters: Axial-Flair, T1- and T2-weighed images; Sagittal T2-weighed images; Coronal-T2-weighed images) revealed calcified granulomas in the left parietal lobe and acute mastoiditis with heterogeneous signal intensity involving the right masticator space with cortical irregularities in the ipsilateral ramus of mandible [Figure 3], [Figure 4], [Figure 5]. Based on clinical and radiological findings, provisional diagnosis of fibrous dysplasia and osteomyelitis were made. An incisional biopsy was taken under local anesthesia from right angle of mandible and sent it for histopathological analysis.
Figure 3: Orthopantomogram showing mixed radiolucency with ground glass appearance along with expansion of the cortex

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Figure 4: Computerized tomography show osteosclerotic lesion with ground glass opacification

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Figure 5: Magnetic resonance imaging showing calcified granulomas in parietal lobe and acute mastoiditis with heterogenous signal intensity alteration in masticator space

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Gross examination of the tissue revealed one greyish-brown soft-tissue specimen measuring 1 cm × 0.6 cm × 0.4 cm in size and multiple firm to hard bony tissue pieces collectively measuring 0.6 cm × 0.4 cm × 0.4 cm in size. Whole specimen kept for routine processing. Hematoxylin and eosin stained section reveals central granuloma formation within which are seen the characteristics colony of microorganisms. These colonies were seen floating in a sea of polymorphonuclear leucocytes. Numerous macrophages were seen around the periphery of the colony. The central area of the colony stained deeply basophilic with a peripheral rim of eosinophilia. Numerous bony trabeculae were also seen. Few of the bony trabeculae were present without osteoblastic rimming and no osteocytes within the bone matrix representing nonvital bone. The connective tissue stroma showed moderate chronic inflammatory cell infiltrate composed of lymphocytes and plasma cells. Areas of hemorrhage with extravasated red blood cells were also noted [Figure 6] and [Figure 7]. In addition, Periodic acid–Schiff stained section confirmed magenta color colonies of microorganisms [Figure 8]. Based on histopathological features, we confirmed the diagnosis of Actinomycotic osteomyelitis.
Figure 6: Colonies of gram-positive bacteria surrounded by purulent exudate (×10)

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Figure 7: Ray fungus like filamentous microorganisms seen surrounded by floating polymorphonuclear leucocytes (×40)

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Figure 8: PAS stained section showing magenta colored colonies of glycogen rich Actinomyces species

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Surgical debridement of the necrotic material and decortication was performed along with Antimicrobial therapy, which includes Amoxiclav 625 mg for 6 weeks and patients is advised for long-term continuous follow-up for 6 months.


  Discussion Top


Actinomycosis is a Greek word comprising of “Aktino” meaning radiating appearance of sulfur granules and “mykos” which labels the condition as mycotic disease. Bollinger described the organism Actinomyces bovis and its ability to cause “lumpy jaw” in cattle.[8] The word Actinomyces depicts “ray fungus,” and based on the general notion that the organism was a fungus.[8] The first person to describe the disease in human was Von Langenback in the year 1848 and endorsed it as fungus. Israel and Wolf in the year 1891 isolated the organism in humans through anaerobic culture at body temperature.[8] Actinomycetes forms a mycelia network of branching filaments like fungus and also thin have cell wall with muramic acid and responds to antibiotic sensitivity like bacteria.[8] Waksman in the year 1960, declared Actinomyces as gram-positive bacteria.[8]

Actinomycosis is a poly-microbial infection and requires other bacteria such as Hemophilus species, anaerobic Streptococci and other Gram-negative bacilli which acts as co-pathogens, provides anaerobic atmosphere for growth of Actinomyces.[9] Since Actinomyces is a weak pathogen and lack tissue decomposing enzyme (hyaluronidase) the co-pathogens aids in achieving pathogenicity.[9] Actinomyces does not invade the mucosal barrier due to its weak pathogenicity.[9] Trauma, Periodontal infections, nonviable teeth and extraction sites are common etiology of this infection.[10] The pathomechanism of the disease is still not clearly elucidated. Actinomyces bacteria are common commensal of the oral cavity and are localized in palatine tonsils, gingival fluid, mucosal surfaces, dentin cavities, and sites of postextraction. It has been hypothesized that the infection manifests especially when the normal microbial flora is altered, and chronic inflammation leads to localized pathological changes in the bone.[11]

Actinomycosis usually affects between 3rd and 6th decade of life, with a definite male predilection (4:1) and mandible being more commonly affected than maxilla.[11] Actinomycosis involving maxilla accounts for only 0.5%–9% of all head and neck cases.[12] It is assumed that the mandibular predominance of the disease owes to the fact that mandible has relatively poor vascularization of the condensed cortical bone.[13] The probable reason for maxilla being a rare site could be attributed to its profuse blood supply.

Clinically, cervicofacial actinomycosis usually involves soft tissues and characteristically manifests as brawny swelling which if untreated discharges through multiple sinuses on to the skin surface.[14] The skin overlying the abscess is purplish red, indurated and has the feel of wood.[14] It is common for the sinus through which the abscess has drained to heal but because of the chronicity of the disease new abscess develop and perforate the skin surface. Thus, the patient over a period of time shows extensive scarring and disfigurement.[14] The infection of the soft tissues may spread through direct extension and it extends to involve the mandible or maxilla.[14]

The diagnosis of Cervicofacial Actinomycosis is made by clinical, radiological, histopathological, and bacteriological examination. Bacterial culture gives a definitive diagnosis. However, bacteriological identification recovery rate is <50% and is quite crucial possibly due to suppressive effect of prior antimicrobial therapy injudiciously used for any possible infection.[11],[15] Therefore, histopathological analysis has been considered as mainstay by many authors.[11],[14],[16],[17] Actinomyces species strongly stain positive for H and E, Periodic acid–schiff, and Giemsa (GMS).[18]

Due to overlapping clinical, radiographic and histopathological features, Actinomycotic osteomyelitis should be differentiated from Fibroosseous Lesion, Benign and Malignant Tumors of Bone, Chronic Osteomyelitis, Chronic granulomatous Lesion such as Tuberculosis, Sarcoidosis and Leprosy and Nocardia in order to achieve a definitive diagnosis. A detailed description of the common and the differentiating features for a conclusive diagnosis is illustrated in [Table 1].
Table 1: Differential diagnosis of actinomycotic osteomyelitis

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Advanced diagnostic methods, such as molecular testing by polymerase chain reaction (PCR) are considered to be an appropriate and promising diagnostic technique.[14] A study was conducted by Hansen et al. identified A. israelii by PCR.[19] Since the study demonstrated reduced sensitivity, they conduced further study and evaluated that increased sensitivity in the result can be obtained if milder decalcifying agents such ethylene di amine tetra acetic acid instead of strong de calcifying agent as trichloroacetic acid are used in bone specimens.[20] Although PCR technique is highly sensitive and specific; cost, availability, and technique sensitization are issues that hamper its routine employment in detection of the microorganism. Even less invasive diagnostic techniques, such as fine needle aspiration are now been studied as effective diagnostic technique for molecular identification.[21] Even after such diagnostic methods, Actinomycosis has been considered as masquerader of the disease;[22]<10% of all infections are correctly diagnosed.[23]

Surgical debridement of the lesion along with antimicrobial therapy and complete resolution of the symptoms is the treatment protocol.[21] Antibiotic sensitivity test used for treatment from proper antibiotic therapy. Penicillin and quinolones are preferred choice of antibiotics.[24] For patients susceptible to beta-lactams, oral amoxicillin is the treatment of choice. Along with amoxicillin, conjunct of metronidazole or beta lactamase inhibitors are used.[25]


  Conclusion Top


Actinomycotic osteomyelitis is a rare disease with nonspecific symptoms and signs making the diagnosis of disease difficult. Early diagnosis can deliver prompt treatment and thus better outcome of the disease. Any suspected clinical lesion should definitely go for incisional biopsy as histopathological diagnosis is the “gold standard” for the disease and false negative result of microbiological cultures may obscure the correct and conclusive diagnosis.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Ting WT, Afshin H, Patey N. Chronic actinomycotic osteomyelitis of the mandible: A case report. J Am Acad Dermatol 2017;76:AB69.  Back to cited text no. 1
    
2.
Park JK, Lee HK, Ha HK, Choi HY, Choi CG. Cervicofacial actinomycosis: CT and MR imaging findings in seven patients. AJNR Am J Neuroradiol 2003;24:331-5.  Back to cited text no. 2
    
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Smith MH, Harms PW, Newton DW, Lebar B, Edwards SP, Aronoff DM. Mandibular actinomyces osteomyelitis complicating florid cemento-osseous dysplasia: Case report. BMC Oral Health 2011;11:21.  Back to cited text no. 3
    
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Brown JR. Human actinomycosis. A study of 181 subjects. Hum Pathol 1973;4:319-30.  Back to cited text no. 5
    
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Bala S, Narwal A, Gupta V, Duhan J, Goel P. Actinomycotic osteomyelitis of mandible masquerading periapical pathology. J Oral Health Comm Dent 2011;5:97-9.  Back to cited text no. 8
    
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Sezer B, Akdeniz BG, Günbay S, Hilmioǧlu-Polat S, Başdemir G. Actinomycosis osteomyelitis of the jaws: Report of four cases and a review of the literature. J Dent Sci 2017;12:301-7.  Back to cited text no. 9
    
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Krzysztofiak A, Deriu D, Roversi M, Grandin A, Cirillo M, Villani A. Actinomycotic osteomyelitis of the jaw in a child. J Oral Health Dent Sci 2019; 3:204.  Back to cited text no. 10
    
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Figueiredo LM, Trindade SC, Sarmento VA, de Oliveira TF, Muniz WR, Valente RO. Actinomycotic osteomyelitis of the mandible: An unusual case. Oral Maxillofac Surg 2013;17:299-302.  Back to cited text no. 11
    
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Nagler RM, Ben-Arieh Y, Laufer D. Case report of regional alveolar bone actinomycosis: A juvenile periodontitis-like lesion. J Periodontol 2000;71:825-9.  Back to cited text no. 13
    
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Baldawa P, Shirol P, Kulkarni D, Koshti S, Mishra N. Actinomycotic osteomyelitis of palate masquerading periapical pathology: A rare case report. Indian J Dent Res 2018;29:247-51.  Back to cited text no. 14
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Yadav SP, Chanda R, Gathwala G, Yadav RK. Actinomycosis of tonsil masquerading as tumour in a 12 year old child. Int J Pediatr Otorhinolaryngol 2002;63:73-5.  Back to cited text no. 15
    
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Yakata H, Nakajima T, Yamada H, Tokiwa N. Actinomycotic osteomyelitis of the mandible: Report of case. J Oral Surg 1978;36:720-4.  Back to cited text no. 16
    
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Peters E, Lau M. Histopathologic examination to confirm diagnosis of periapical lesions: A review. J Can Dent Assoc 2003;69:598-600.  Back to cited text no. 17
    
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Chandler FW, Watts JC. Fungal diseases. In: Damjanov I, Linder J, editors. Anderson's Pathology. 10th ed. St. Louis: MO: Mosby-Year Book; 1989. p. 951-82.  Back to cited text no. 18
    
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Hansen T, Kunkel M, Kirkpatrick CJ, Weber A. Actinomyces in infected osteoradionecrosis-underestimated? Hum Pathol 2006;37:61-7.  Back to cited text no. 19
    
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Hansen T, Kunkel M, Springer E, Walter C, Weber A, Siegel E, et al. Actinomycosis of the jaws-histopathological study of 45 patients shows significant involvement in bisphosphonate-associated osteonecrosis and infected osteoradionecrosis. Virchows Arch 2007;451:1009-17.  Back to cited text no. 20
    
21.
Volante M, Contucci AM, Fantoni M, Ricci R, Galli J. Cervicofacial actinomycosis: Still a difficult differential diagnosis. Acta Otorhinolaryngol Ital 2005;25:116-9.  Back to cited text no. 21
    
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Rankow RM, Abraham DM. Actinomycosis: Masquerader in the head and neck. Ann Otol Rhinol Laryngol 1978;87:230-7.  Back to cited text no. 22
    
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Weese WC, Smith IM. A study of 57 cases of actinomycosis over a 36-year period. A diagnostic 'failure' with good prognosis after treatment. Arch Intern Med 1975;135:1562-8.  Back to cited text no. 23
    
24.
Tanaka-Bandoh K, Watanabe K, Kato N, Ueno K. Susceptibilities of actinomyces species and propionibacterium propionicus to antimicrobial agents. Clin Infect Dis 1997;25 Suppl 2:S262-3.  Back to cited text no. 24
    
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]
 
 
    Tables

  [Table 1]



 

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