Jaw actinomycosis—An opportunistic infection: Case series

Abstract

Jaw actinomycosis is a quite rare invasive facultative bacterial infection caused by Actinomyces, Gram-positive filamentous bacilli found in human commensal. A break in continuity of epithelium due to surgery, trauma or previous infection can lead to deeper invasion of bacteria causing infection. The risk factors for actinomycosis are trauma, caries, debilitation, and poorly controlled diabetes mellitus. Clinical presentation can mimic other pathologies, such as fungal infection tuberculosis, granulomatous diseases, so the diagnosis of actinomycosis is delayed or misdiagnosed. For the definitive diagnosis of jaw actinomycosis, medical history, dental history histopathological examinations and microbiological culture are important parameters. Actinomycotic bacteria are sensitive to antibacterial agents hence chemotherapeutic agents are used for treatment. This report presents case series of jaw actinomycosis involving mandible and maxilla. The final diagnosis was supported by histopathology.

INTRODUCTION

Actinomycosis is quite a rare invasive bacterial infection caused by Actinomyces, which are gram-positive filamentous bacilli found in the human commensal flora of the oropharynx, gastrointestinal tract, and urogenital tract.[1] Common species causing cervicofacial actinomycosis is Actinomyces israelii but other species like Actinomyces naeslundii, Actinomyces viscous, and Actinomyces odontolyticus can cause infection. It is a polymicrobial infection, for the infectivity presence of companion bacteria like anaerobic streptococci, fusiform or gram-negative bacilli, and Haemophilus species are needed. Whenever there is a break in continuity due to surgery, trauma or previous infection can lead to a deeper invasion of bacteria causing infection.[2]

Clinical presentation can mimic other pathologies, such as nocardiosis fungal infections, active Mycobacterium tuberculosis infection, or other granulomatous diseases, so the diagnosis of actinomycosis is delayed,[2] and may be misdiagnosed. Less than 10% of cases of head-and-neck diseases are correctly diagnosed and may prove fatal in up to 28% due to misdiagnosis.[3] For the definitive diagnosis of actinomycoses, radiography, medical history, histopathological examinations and microbiological culture are important parameters.[4] Actinomycotic bacteria are sensitive to antibacterial so chemotherapeutic agents are used for treatment.[2]

The purpose of this report is to present a case series of osteomyelitis with actinomycosis.

DISCUSSION

Actinomyces are non-sporing, anaerobic gram-positive bacteria, belonging to the Actinomycetales order. There are 49 different species of the Actinomyces genus, out of which 27 species are causative agents in human infections.[4] Actinomyces is a commensal bacterium in the oropharynx, gastrointestinal tract, and female genital tract in human beings and breaks in mucosal continuity cause diseased conditions.[3] In humans, it was first recognised by Von Langenbeck in 1845.[5]

According to the site, they are classified as orocervicofacial (40%–60%), abdominopelvic (20%–30%), and thoracic (20%–25%) among cervicofacial is the most common type.[3,5]

The causes of actinomycosis infection are extraction site trauma, periodontal infection, non-viable teeth, diabetes, immunosuppression, corticoid treatment for extended periods, alcoholism, and smoking and disease recurrence may be due to incomplete response to antimicrobial agents.[6,7]

Actinomyces are devoid of hyaluronidases, which is an enzyme essential for tissue decomposing so, they need other additional bacteria like streptococci and staphylococci for their pathogenicity.[8]

The most common age group affected is 30–60 years with male predilection (4:1).[7] Similar findings were found in our cases. Clinically. It shows woody swelling with pain (lumpy jaw), and suppuration with the formation of fistulae or sinus tracts with 'sulphur granules', which is a characteristic diagnostic marker of Actinomyces. But in 50% of cases, these are not found because of prior antibiotic course and long-standing infections in these series also sulphur granules were not demonstrated.[3] In our series, almost all cases showed pus discharge, exposed bone in one and pain in one case [Table 1, Figures 1 and 2].

Table 1.

Clinicopathological parameters of cases

Entity	Case 1	Case 2	Case 3	Case 4
Clinical features [Figures 1 and 2]	Age/sex: 40/M D/H: extraction with 16 E/O: swelling seen on right zygomatic region I/O pus discharge from 13-15. Mobility with 13-15	Age/sex: 65/M D/H extraction with 42 and 43. I/O: Sinus and pus discharge with 41-43	Age/sex: 65/F D/H: extraction 16. E/O: swelling seen on right zygomatic region I/O: pus discharge and exposed bone with 16	Age/sex: 42/M D/H: I/O: pus discharge and pain with 31-43 region
Radiological findings [Figure 3]	Periapical radiolucency was seen with 13-15	Periapical radiolucency was seen with 33-36	Radiolucency was seen in the right maxillary region	Radiolucency was seen with 35-46
Histopathological findings [Figure 4]	Clumps of basophilic radiating filaments with peripheral eosinophilic bulb, which surrounded by chronic inflammatory cell infiltrate is evident suggestive of actinomycotic colonies	Clumps of basophilic radiating filaments with a peripheral eosinophilic bulb, which surrounded by chronic inflammatory cell infiltrate is evident suggestive of actinomycotic colonies	Clumps of basophilic radiating filaments with a peripheral eosinophilic bulb. which surrounded by chronic inflammatory cell infiltrate is evident suggestive of actinomycotic colonies	Clumps of basophilic radiating filaments with a peripheral eosinophilic bulb, which surrounded by chronic inflammatory cell infiltrate is evident suggestive of actinomycotic colonies
PAS staining [Figure 5]	Positive	Positive	Positive	Positive

M/H=medical history, D/H=dental history, E/O=extraoral examination, I/O=intraoral

Figure 1.

Extraoral examination

Figure 2.

Intraoral examination

Radiographically, they can be useful for the recognition of extension in bone but are nonspecific for actinomycosis.[7] It may show radiolucency with hazy and fuzzy bony trabeculae or diffuse irregular sclerosis of the bone, often described as a 'cotton-wool' appearance.[8] These series show irregular and patchy radiolucent areas with affected areas [Table 1 and Figure 3].

Figure 3.

Radiological examination

In the present case, clinical and radiographic features provided a provisional diagnosis of osteomyelitis.

Bacteriological and histopathological examination is required for the final diagnosis of actinomycosis,[8] but the bacterial isolation from culture, the success rate is less than 30%, because actinomyces require a careful anaerobic culture of these bacteria that are sensitive to oxygen; up to 14 days of strict anaerobic incubation.[3,7] Prior antibiotic treatment, overgrowth of organisms such as Aggregatibacter species, Bacteroides and Fusobacterium.[3] All the patients in the series have a long-standing infection and they had taken antibiotics for the same so laboratory cultures were not attempted.

Histopathological findings of incisional biopsy can help greatly in the diagnosis of actinomycosis. The most common microscopic feature of actinomycosis is the central zone of necrosis containing multiple basophilic granules that represent lobulated microcolonies of Actinomyces with an outer zone of granulation. In histopathological staining, these colonies appear as round or oval basophilic masses with an eosinophilic terminal.[9,10] In this series, observed features are clumps of basophilic radiating filaments with a peripheral eosinophilic bulb surrounded by chronic inflammatory cell infiltration, which is evidence suggestive of actinomycotic colonies [Table 1 and Figure 4]. PAS staining shows magenta-pink-coloured radiating filaments with a peripheral bulb [Table 1 and Figure 5].

Figure 4.

Histopathological examination under high magnification

Figure 5.

PAS staining

Treatment for actinomycotic infection includes abase drainage, and surgical debridement, and if a bone is involved, curettage and ablating sequestra are required with antibiotic therapy like penicillin, erythromycin, tetracycline, clindamycin, imipenem, streptomycin, and cephalosporin. In this series, patients received the same treatment regimen.[11]

The prognosis of actinomycosis was poor before the antibiotic treatment era but nowadays prognosis is good with different effective antibiotics.[10,11]

CONCLUSION

The diagnosis of actinomycosis in orofacial soft tissues is often challenging and appropriate diagnosis greatly influences the prognosis of cervicofacial actinomycosis. A clinical, cultural and histopathological examination may provide more valuable information. For a good prognosis, a multidisciplinary approach with antibiotic therapy and surgical management is required.

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.