Journal of Indian Academy of Oral Medicine and Radiology

CASE REPORT
Year
: 2021  |  Volume : 33  |  Issue : 4  |  Page : 487--489

Actinomycotic osteomyelitis of maxilla in a diabetic patient- “A rare case report & review of literature”


Basavaraj T Bhagawati1, Manisha Solanki2, Hemlata Solanki2, Manish Sharma3,  
1 Department of Oral Medicine and Radiology, Surendera Dental College and Research Institute, Rajasthan, India
2 Department of Oral and Maxillofacial Surgery, Surendera Dental College and Research Institute, Rajasthan, India
3 Department of Oral and Maxillofacial Pathology, Surendera Dental College and Research Institute, Rajasthan, India

Correspondence Address:
Dr. Basavaraj T Bhagawati
Department of Oral Medicine and Radiology, Surendera Dental College and Research Institute, Rajasthan
India

Abstract

Actinomycosis may be a relatively rare infection caused by saprophytic bacteria of the mouth and alimentary canal which will become pathogenic. The chronic hyperglycemia in Diabetes Mellitus induces events that promote structural changes in various tissues and are related to problems in wound healing. This infection remains largely unknown to most clinicians due to its different presentations, and palatal involvement is extremely rare. This report describes the case of a 50-year-old man who was diagnosed with actinomycosis involving mucosal surface. The main clinical, histopathologic, and therapeutic characteristics and diagnosis of actinomycosis are reviewed. Till date, 4 cases of actinomycosis involving the palatal surface are reported.



How to cite this article:
Bhagawati BT, Solanki M, Solanki H, Sharma M. Actinomycotic osteomyelitis of maxilla in a diabetic patient- “A rare case report & review of literature”.J Indian Acad Oral Med Radiol 2021;33:487-489


How to cite this URL:
Bhagawati BT, Solanki M, Solanki H, Sharma M. Actinomycotic osteomyelitis of maxilla in a diabetic patient- “A rare case report & review of literature”. J Indian Acad Oral Med Radiol [serial online] 2021 [cited 2022 Jun 26 ];33:487-489
Available from: https://www.jiaomr.in/text.asp?2021/33/4/487/333866


Full Text



 Introduction



Actinomycotic osteomyelitis of maxilla is comparatively rare in comparison to mandible, possibly due to rich blood supply and porous bone which provides increased oxygen supply.[1],[2],[3],[4] The primary actinomycosis arising within the maxilla with contiguous involvement of para-nasal sinus with development of oroantral fistula and palatal perforations is rare.[3],[4],[5],[6],[7] It's a myriad of clinical presentations and may be a potentially benign and entirely curable disease but if not noticed early, has the propensity to wreck extensively. This unusual case shows chronic actinomycotic osteomyelitis of maxilla with suppurative and sclerosing features in a 50-year-old uncontrolled diabetic male patient.

 Case Report



Clinical history

A 50-year-old male patient presented with a complaint of unhealed extraction site of 26 & 27 region. He had a previous history of intra oral swelling in the same region one and half year back which was painful with pus discharge, gradual loosening and self-exfoliation of 26 and 28, six months back. He took medications & incision and drainage was done by local dentist but pain was not relieved. Patient had history of numbness since last 3 months on left side of face. He was known case of diabetes since last 10 years and was on irregular oral hypoglycemics.

Clinical findings

On clinical examination, there were no significant extraoral findings. On Intraoral examination, an asymmetrical necrotic defect measuring about 2-2.5 cm approx. anteroposteriorly at 27, 28 regions was seen [Figure 1]. Oroantral communication and pus discharge was present at 27-28 region. 11,12,13,21,22,23,24,25 teeth were non-vital. Infected site was non-tender, palate was soft to firm in consistency. Based on the history and clinical findings, provisional diagnosis of chronic Osteomyelitis was made.{Figure 1}

The actinomycosis was given as differential diagnosis.

Diagnostic asessment

The patient was advised Orthopantomogram(OPG), Computed Tomogram (CT) scan and a routine hemogram. The OPG showed an aggressive destructive lesion involving palate. Axial view of CT scan reveals an extensive mixed density lesion involving the left alveolar process of maxilla with a moth-eaten appearance [Figure 2]. Complete blood count (CBC) showed neutrophilic leucocytosis, Raised Random blood sugar (RBS), HbA1c and ESR.{Figure 2}

Therapeutic intervention & follow up

Considering diabetic condition, age and size of the lesion case was operated under G.A where Sequestrectomy and debridement along with extraction of 24 & 25 teeth and tissue was sent for histopathological examination. Dehiscence and wound opening were found in the same area. Biopsy report was suggestive of Actinomycotic Osteomyelitis [Figure 3]. He was treated with IV penicillin for 14 days along with IV clindamycin, for 7 days. Later the patient was given oral clindamycin for 6 weeks. The patient reported after one month for follow up and found improvement with resolution of the lesion. The patient was followed up on monthly basis.{Figure 3}

 Discussion



Actinomycosis of maxilla reports only 0.5–9% of cases among maxillofacial region.[4],[5] However, the precise mechanism of actinomycotic osteomyelitis is not yet found, it's found that inflammation begins as soon as after the normal commensals of oral cavity is disturbed. It causes sclerosing sort of osteomyelitis resembling bone tumors.[2] Actinomycosis is a relatively rare infection caused by saprophytic bacteria of the oral cavity and gastrointestinal tract that can become pathogenic.[8]

Actinomyces species are saprophytic bacteria of the oral cavity and gastrointestinal tract. The bacteria exhibit a low degree of virulence and are commonly found in the saliva and in dental plaque. However, under certain circumstances that compromise anatomic barriers and host susceptibility, their pathogenic form can cause actinomycosis. Actinomycotic Osteomyelitis has been associated with multiple systemic diseases like diabetes, autoimmune disorders, malignancies, malnutrition and acquired immunodeficiency syndrome.

The clinical presentation of multiple pus discharging sinuses is typical of actinomycotic infection, which is ruled out after culture of the pus sample. The actinomycotic infection forms external sinuses that discharge distinctive sulfur granules and spreads unimpeded by anatomical barriers. Nearly every structure and space of the head and neck may be the initial site of involvement. Males are more often affected than women, with a 3:1 ratio.[8]

The osteomyelitis of maxilla with involvement of sinus might be a result of adjacent oral soft tissue involvement because of periapical or periodontal infection in present case. The chronic purulent infection, poor oral health with unhealed extraction sites, and surgical maneuvers within this case presents the infiltration site of those organisms into deeper layers was from gingiva, periodontal tissues, or chronic periapical abscess and oral surgical procedures extended the pathogenicity of those organisms.[5],[6] Marx et al.[9] labelled it as mutualism than synergism because sulfur granules are formed in tissues but not under laboratory conditions signifying the importance and role of associate bacteria in colony formation, growth, and evasion of host defenses mechanism.

The oral antibiotic course is typically less productive as found within this case and will be used along with surgical procedures. In chronic long-standing cases, fibrosis can progress into high level sclerosis; endosteal sclerosis can cause therapeutic failure as reduced vascularity inhibits penetration of antibiotics. The thickness of sulfur granule colony, protective nature of sulfur granule, and therefore the characteristic necrosis and fibrosis surrounding such infections need large diffusion gradients, making high doses a requirement.[1],[2],[9] Treatment should be vigorous with removal of granulation and resection of sequestrated bone till healthy tissue is exposed. The drug of choice is penicillin and clindamycin with duration ranging from 3 to 12 months.[1],[2],[10]

Diagnosis is usually delayed due to varied presentations and that they are cultured in less than 50% of cases and are the primary element of diagnosis in less than 10% of cases.[2] The sulfur granules aren't pathognomonic as they're produced by mycotic pathogens, Nocardia, Botryomycosis and since such cases are rare, Myerowitz states that the presence of sulfur granules virtually guarantees the diagnosis of actinomycosis.[2] A clinical diagnosis could be difficult and various problems are related to bacteriological culture but histopathology is gold standard. The nucleic acid probes and polymerase chain reaction methods are developed for fast and precise identification but are highly expensive.[5]

 Conclusion



Oral mucosa is usually the penetration site of Actinomyces into deeper tissues. Histopathology remains reliable diagnostic aid. Although the cases of actinomycosis of maxilla are rare, it should be included in the one of the differential diagnosis of osteomyelitis.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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