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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 10  |  Issue : 2  |  Page : 113-116

An unusual case of intraoral actinomycosis: Case report and literature review


Department of Pathology, 158 Base Hospital, Darjeeling, West Bengal, India

Date of Submission30-May-2020
Date of Decision05-Oct-2020
Date of Acceptance16-Oct-2020
Date of Web Publication16-Feb-2021

Correspondence Address:
Dr. Hemant Kumar Sharma
Department of Pathology, 158 Base Hospital, Bagdogra, Darjeeling - 734 424, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijohs.ijohs_19_20

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  Abstract 


Actinomycosis is an infrequent chronic suppurative granulomatous disease caused by commensal bacteria of Actinomyces species. It usually affects the orocervicofacial region and classically presents with chronic abscess or persistent discharging sinus tract formation along with “sulfur granules” and woody fibrosis. Bacteriological and histopathological examination confirming the presence of Actinomyces colonies is essential for diagnosis and treatment modalities to include long-term oral and intravenous antibiotics. We report a challenging case of a 38-year-old male who presented with pain, swelling, and numbness around the left upper jaw after undergoing dental extraction about 1 month back. No symptoms of discharging pus, sinus tract, or abscess formation or fibrosis were seen. Based on the clinical history and symptoms, a presumptive diagnosis of intraoral actinomycosis was made, but no organism could be cultured. Finally, surgical resection was done, and Actinomyces involving the first and second molar teeth of the upper left maxilla was confirmed on histopathology. Intraoral actinomycosis requires long-term antibiotic treatment, but in our patient, a newer treatment modality was attempted which yielded excellent results without any recurrence after 1 year.

Keywords: Actinomyces, cervicofacial actinomycosis, oral cavity, sulfur granules


How to cite this article:
Sharma HK, Prashar S. An unusual case of intraoral actinomycosis: Case report and literature review. Int J Oral Health Sci 2020;10:113-6

How to cite this URL:
Sharma HK, Prashar S. An unusual case of intraoral actinomycosis: Case report and literature review. Int J Oral Health Sci [serial online] 2020 [cited 2021 Mar 8];10:113-6. Available from: https://www.ijohsjournal.org/text.asp?2020/10/2/113/309443




  Introduction Top


In 1845, Langenbeck first described actinomycosis in humans as a rare chronic suppurative granulomatous lesion caused by endogenous commensal organisms of genus Actinomyces.[1],[2] Out of more than 49 species, Actinomyces israelii (52%) is the most frequently isolated species followed by A. Naeslundii, A. Viscosus, A. Meyeri, and A. Odontolyticus in remaining cases.[1],[2],[3],[4] Earlier thought to be fungi, microbiological characteristics of these organisms proved them to be slow-growing, Gram-positive anaerobic or microaerophilic nonspore-forming, nonacid fast filamentous branching bacteria.[1],[4],[5],[6]

Due to its rarity, clinical presentation similar to various other conditions including malignant neoplasm, and difficulty in culture and isolation, Actinomyces is often described as a great masquerader among head-and-neck diseases as only <10% cases are correctly diagnosed and may prove fatal in up to 28% misdiagnosed cases.[3],[4] Herein, we report a challenging case of intra-oral actinomycosis who presented with atypical presentation and treated with newer treatment modality to increase awareness about this infrequent entity.


  Case Report Top


A 38-year-old male patient reported to a dental surgeon with the chief complaint of pain in his upper left back region of the jaw and numbness in the chin for the past 10–12 days. His symptoms started after he underwent extraction of the upper left 26 and 27 (first and second molar teeth) in a clinic about a month back. After extraction, he was prescribed broad-spectrum antibiotics and painkillers.

Two weeks after extraction, he returned with excruciating pain in the same region and numbness in the chin for the previous 5 days and was subsequently referred to our dental center. His had no past or family medical history of diabetes, hypertension, or renal diseases. He was a chronic smoker (1–2 bidi packets/day) and alcoholic (2–4 pegs/day) for the last 10 years. His extraoral examination was unremarkable, whereas intraoral examination revealed missing 26 and 27 in the upper left maxilla with the presence of an unhealed extraction socket filled with slough and debris and exposed buccal and lingual cortices. On palpation, the lesional area was tender, edematous, soft to firm in consistency, and immobile. However, no trismus, discharging sinus tract formation, or fibrosis was seen [Figure 1].
Figure 1: Orthopantogram showing generalized bone loss and scooped-out radiolucent lesion with respect to the 26 and 27 teeth (blue arrow)

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His routine blood investigations showed normal complete hemogram except an elevated erythrocyte sedimentation rate, mild leukocytosis (total leukocyte count 14,500/mm3, differential leukocyte count P83, L15, E01, M01), and raised C-reactive protein levels. On diabetic workup, his fasting (95 mg/dL) and postprandial (137 mg/dL) blood sugar levels were within normal limits. On radiographic examination, a well-circumscribed scooped-out or saucer-shaped radiolucent area surrounded by a sclerotic border extending from 26 to 27 in the left maxilla was noticed. The surrounding area appeared unremarkable [Figure 1]. Based on his clinical history, symptoms, and radiological evidence, a provisional diagnosis of osteomyelitis was made and the lesion was subjected to excisional biopsy under local anesthesia.

Histopathological examination of hematoxylin and eosin-stained section showed central suppurative necrotic area and presence of numerous hematoxyphilic filamentous structures surrounded by an eosinophilic material in a background of polymorphous diffuse inflammatory cell infiltration. Gram staining of the tissue showed these basophilic filamentous structures to be Gram-positive elements suggestive of actinomycotic infection [Figure 2],[Figure 3],[Figure 4]. Therefore, a final diagnosis of actinomycotic osteomyelitis of the maxilla was offered. The patient was initially treated with a combination of intravenous penicillin and metronidazole followed by oral amoxicillin 500 mg three times a day for 21 days. He was followed up for 6 months and has shown tremendous clinical improvement and complete healing without any recurrence so far.
Figure 2: Connective tissue stroma with multiple deeply basophilic actinomycotic colonies (sulfur granules) shown by yellow arrow, surrounded by necrosis and inflammatory infiltrate (H and E, ×200)

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Figure 3: Club-shaped basophilic colonies of filamentous organisms surrounded by inflammatory infiltrate (yellow arrow, H and E, ×400)

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Figure 4: Gram stain shows colonies of Gram-positive filamentous organisms (yellow arrow, Gram stain ×200)

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  Discussion Top


In humans, Actinomyces exist as commensal bacteria in the oropharynx, gastrointestinal tract, and female genital tract.[2] Due to the low virulence potential of these organisms, a breach in the mucosal barrier and subsequent spread to subcutaneous tissue are essential prerequisite for pathogenesis and appearance of clinical symptoms.[4]

Based on their site of occurrence, Cope, in 1938, classified actinomycosis into the following three types: orocervicofacial (40%–60% of cases), abdominopelvic (20%–30% of cases), and thoracic (20%–25% of cases) actinomycosis.[2],[3],[4] Usually, orocervicofacial actinomycosis affects middle-aged males aged between 40 and 50 years (M:F ratio 3:1) who are chronic smoker or alcoholic, have impaired immunity and poor oral hygiene, undergone invasive dental procedures, or suffered orofacial traumas.[1],[2],[3],[4],[5]

Intraorally, it is usually isolated from dental plaque, dental caries, tonsillar crypt, gingival crevices, and periodontal pockets.[2] It affects mandible more than maxilla (4:1 ratio) and initially presents with fever (>50% cases) followed by chronic abscess or persistent discharging sinus tract formation (40% cases) along with “sulfur granules” and woody fibrosis, giving a hardboard-like feel to the jaw and hence the name “Lumpy jaw.”[1],[2],[3],[4],[5],[6]

Sulfur granules, named so due to their yellowish color and macroscopic resemblance to sand grains, are considered important diagnostic markers of Actinomyces species but rarely seen these days due to frequent use of antibiotics.[2],[6] They measure up to 1 mm in diameter and predominantly seen in the central necrotic area.[2] Their mechanism of formation is by deposition of calcium phosphate in masses of filaments of bacteria due to phosphatase activity from host tissue inflammation and predominantly seen in the central necrotic area.[1],[2] However, they are not pathognomonic of actinomycosis as similar granules may be observed in other bacterial infections (Staphylococcus, Actinobacillus lignieresi, Nocardia asteroides/brasiliensis, and Streptomyces madurae infections) and mycotic infections (Sporotrichum and Phialophora) and noninfectious processes like around inert material, allergic conjunctival granuloma, and hypereosinophilic syndrome.[1],[2],[6] In addition, even in about 50% of proven cases of actinomycosis, sulfur granules are not seen probably due to prior course of antibiotics and long duration of illness.[2] In our patient also, sulfur granules could not be demonstrated and can be attributed to similar reasons.

Diagnosis necessitates direct isolation of Actinomyces from a clinical specimen or sulfur granules and confirmation on cultures and histopathology.[1],[2],[3],[4],[5],[6] However, the success rate for isolation via culture is <30% owing to fastidious growth requirements requiring up to 14 days of strict anaerobic incubation, previous antibiotic treatment, or aerobic and anaerobic overgrowth of organisms such as Bacteroides, Fusobacterium, or Aggregatibacter species.[3],[4],[6],[7] In our patient, due to the presence of established clinical findings, history of infection, and previous course of antibiotics, laboratory culture was not attempted. Instead, the decorticated tissue was sent for histopathological examination which confirmed the diagnosis. Infection usually spreads by direct contiguous spread to adjacent muscles and bones and subsequently to the trachea, thorax, and central nervous system without any regard to anatomic planes or lymphatic drainage.[1],[2],[3],[4],[5]

Its main differential diagnosis includes bacterial abscess, tuberculosis or nocardiosis, brachial cyst, sialadenitis, and primary or metastatic neoplasm such as squamous cell carcinoma.[2],[6],[7] Conventional treatment options include long-term oral and intravenous antibiotic administration followed by surgical resection if the patient does not respond to medical treatment or there is extensive necrosis, sinus tracts, and fistulas due to which antibiotics cannot penetrate. The standard antibiotic regime includes intravenous penicillin G over 2 to 6 weeks followed by oral penicillin V depending on clinical improvement.[5],[6] In patients who are allergic to penicillin, other antibiotics such as tetracycline and erythromycin can also be used.[2],[4]

Recently, Moghimi et al. in their retrospective analysis and review of 19 cases postulated that surgical resection in combination with intravenous penicillin and metronidazole while awaiting clinical improvement and subsequently, oral antibiotics for 2–4 weeks are generally very efficient.[6] Nagler et al. also hypothesized that due to compartmentalization of the Actinomyces within the necrotic granulation tissue, antibiotics cannot reach and act properly on them, leading to a long duration of treatment. Hence, before starting the antibiotic treatment, curettage or lesional resection facilitates their penetration and shortens the duration of treatment.[8] Due to the extensive necrotic area, our patient was also treated with a combination of surgical debridement and antibiotics and showed tremendous clinical improvement for the last 1 year without any recurrence so far.


  Conclusion Top


Atypical features of our case were the following:

  1. Missing classical presentation of an abscess or sinus tract formation, woody fibrosis, and sulfur granules
  2. No medical history of impaired immunity
  3. Limited site involvement without any contiguous spread to adjacent tissue; and
  4. Use of a combination of surgery and medical management.


Therefore, this report aimed to stress upon the fact that orofacial actinomycosis can present atypically in the absence of classical symptoms and along with definitive and timely diagnosis, a combination of surgical and conservative treatment is necessary for optimal recovery.

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.



 
  References Top

1.
Rathnaprabhu V, Rajesh R, Sunitha M. Intraoral actinomycotic lesion: A case report. J Indian Soc Pedod Prev Dent 2003;21:144-6.  Back to cited text no. 1
[PUBMED]    
2.
Gajdács M, Urbán E, Terhes G. Microbiological and clinical aspects of cervicofacial Actinomyces infections: An overview. Dent J (Basel) 2019;7:85.  Back to cited text no. 2
    
3.
Gaweda A, Wojciechowicz J, Barszczewski G, Tomaszewski T. Cervicofacial actinomycosis following jaw pathology-case reports. J Pre-Clin Clin Res 2016;10:73-5.  Back to cited text no. 3
    
4.
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. 4
    
5.
Thukral R, Shrivastav K, Mathur V, Barodiya A, Shrivastav S. Actinomyces: A deceptive infection of oral cavity. J Korean Assoc Oral Maxillofac Surg 2017;43:282-5.  Back to cited text no. 5
    
6.
Moghimi M, Salentijn E, Debets-Ossenkop Y, Karagozoglu KH, Forouzanfar T. Treatment of cervicofacial actinomycosis: A report of 19 cases and review of literature. Med Oral Patol Oral Cir Bucal 2013;18:e627-32.  Back to cited text no. 6
    
7.
Jangla SM, Machhi BS, Bhandarkar PD. Invasive actinomycosis of maxilla-an unusual case report. J Krishna Inst Med Sci 2019;8:90-3.  Back to cited text no. 7
    
8.
Nagler R, Peled M, Laufer D. Cervicofacial actinomycosis: A diagnostic challenge. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;83:652-6.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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