Rare actinomycosis limited to the bone of the mandible.
Actinomycosis is an uncommon chronic infectious disease caused by Bacterium Actinomyces Israelii, which is an anaerobic gram positive oral flora that may become pathologic due to poor oral hygiene, dental/periodontal problems, trauma or following oral surgical procedures. (1, 2) It is usually found in tonsillar crypts, gingival cervices, carious lesions, and non-vital dental root canals. (2,3) Because the oral mucosa is often portal of entry into deeper tissues, actinomycosis localized to the bone is extremely unusual. (4) Actinomycosis consists of three different forms: cervicofacial (the most common form), abdominal and pulmono-thoracic. (1, 2) Actinomycosis is diagnosed with positive culture or detection of actinomyces colonies and sulphur granules in histopathological specimens. (1)
A 13 years old girl was referred to Gazi University Faculty of Dentistry Department of Oral and Maxillofacial Surgery with a history of tenderness over the right mandibular premolar region. The tooth was vital with no past history of caries, fracture, or trauma. She had no cervical and submandibular lymphadenopathy. The dental history of the patient documented scaling and root planing three weeks ago. Radiologic examination revealed a lyitic lesion 1x1cm in diameter at the middle third of second mandibular premolar. Some lyitic lesions (cryptic foci) were observed at the right and left mandibular canine without mucosal swelling (fig 1). In fine needle aspiration dense yellow to green aspirate was obtained. Under local anesthesia the lesions was completly excisted and sent to oral pathology department for evaluation. Postoperatively, the patient was prescribed amoxicillin 500 mg orally three times a day for 1 week. The post-operative healing was uneventful.
The gross specimen was of homogeneous white appearance and rubbery consistency. The center of the specimen had purulent exudate.Microscopically granulomatous inflammation with a mixed type of inflammatory cell infiltration had been observed. In the center of this granulomatous structure, an actinomycotic colony (sulfur granules) with a radial pattern was noted (fig 2). The periphery of the lesion was intensely fibrotic and the colonies were gram positive (Brown Brenn stain) (fig 3). The final diagnosis was cervicofacial actinomycosis, the abdominal and thoracic types of disease were not detected. 1000 mg of the same antibiotic twice a day for 6 months was prescribed. Complete improvement of the lesions was observed after 5 weeks.
Actinomycosis is an indolent, slowly progressive infection which usually affects cervicofacial, thoracic and abdominal tissues. (3) Actinomycosis is especially seen in middle aged men and is rare in children. In the cervicofacial region, poor oral hygiene and odontogenic infections have been suggested as the main source of actinomycosis. (6)
Actinomycosis is an infectious disease that classically presents as an acute or chronic episode. Chronic actinomycosis is slowly progressive, painless, indurated, which can produce multiple abscesses and cutaneous fistulas. The acute and less frequent form is a rapidly progressive, tender and fluctuant mass, suggestive of an acute dental infection. Actinomycosis should be suspected when sulfur-like granules are observed in pus drainage. (2) In the present case the absence of any draining sinus formation may be explained by the use of potent antibiotics at different time intervals.
Actinomycosis of bone is a rare disease that usually result from adjacent soft tissue infection, but may be also associated with trauma, infected osteoradionecrosis, bisphosphonate-associated osteonecrosis and invation of microorganisms throught out periodontal ligament. (6) In this patient non of the known reasons have been identified except our suspeciuos of unsteril scaling and root planing.
Sulfur granules are thought to be characteristic of actinomycotic infection but they reported only in 40% of the cases. (8, 9) These granules represent colonies of bacteria but they are not pathognomic of actinomycosis, because they are also produced by other groups of bacteria and fungi such as Staphylococcus, Actinobacillus lignieresi, Sporotrichum and Phialophore. (8)
It's pathogenesis is related to its ability to act as an intracellular parasite that resist phagocytosis. Lymph node involvement is not a feature, unless contigious nodes are involved directly. (5) Oral infection may spread to the tongue, hypopharynx, larynx and paranasal sinuses. (10)
Because of the decrease in vascular supply, the penetration of antibiotics to the lesion is difficult. Therefore, the lesion should be removed surgically and the surrounding tissues should be intensely debrided (11). Following surgery, long term antibiotic therapy should be prescribed. Hyperbaric oxygen usage as an adjanctive therapy have also been reported. (8)
Actinomycosis is a very rare infectious disease in children. Due to the opportunistic characteristics of the actinomycotic infection, early and adequate differential diagnosis prior to therapeutic attempts, as well as management steps are of great importance in the oral cavity to prevent the spread of the disease.
(1.) Ozcan C, Talas D, Gorur K, Aydin O, Yildiz A. Actinomycosis of the middle turbinate: an unusual cause of nasal obstruction. Eur Arch Otorhinolaryngol. Epub 2004 Nov 12. 2005 May;262(5):412-415.
(2.) Regezi JA, Sciubba J, Jordan R. eds. Oral pathology: clinicalpathologic correlations. 4nd ed. Philadelphia: Saunders, 2003; 33-34
(3.) Topazian Richard G., Goldberg Morton H. Hupp James R Oral and Maxillofacial InfectionsSaunders; 4th edition pg 231-234.
(4.) Pindborg jj. Atlas de enfermedades de la mucosa oral, 4th ed. Barcelona: Savlat, 1986: 28.
(5.) Lerner P I, Actinomyces and Arachnia Species. In: Mandell G L, Bennett J E and Dolin R, Editors, Mandell, Douglas and Bennett's principles and practice of infectious diseases, Churchill Livingstone (1988), pp. 1427-1433.
(6.) Russo, TA.: Agents of Actinomycosis. In: Mandell, GL., Bennett, JE. and Dolin, R., Eds. Principes and Practice of Infectious Diseases, 6 th Ed., 2005 p.2924-2934
(7.) Binford CH, Connor DH, eds. Pathology of Tropical and Extraordinary Diseases. Washington, DC: Armed Forces Institute of Pathology; 1976:552-554
(8.) Miller M, Haddad AJ.Cervicofacial actinomycosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. Review 1998 May;85(5):496-508.
(9.) Hirshberg A, Tsesis I, Metzger Z, Kaplan I. Periapical actinomycosis: a clinicopathologic study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2003 May;95(5):614-620.
(10.) Brown JR.Human actinomycosis: a study of 181 subjects. Hum Pathol 1973; 4: 319-330.
(11.) Ogutcen-Toller M, Alkan A, Baris S, Cakir N. An unusual form of Actinomycosis of the mandible with a resultant gross sequester in a 4-year-old child: a case report.
Benay Yildirim  and Alaa M. Shuybat 
 Assistant Prof.
 PhD Candidate
Gazi University, Faculty of Dentistry Department of Oral Pathology, Ankara-TURKEY
Received: October 14, 2012;
Review Completed: November 12, 2012;
Accepted: December 11, 2012
Available Online: March, 2013 (www.nacd.in)
Email for correspondence: firstname.lastname@example.org
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|Title Annotation:||CASE REPORT|
|Author:||Yildirim, Benay; Shuybat, Alaa M.|
|Publication:||Indian Journal of Dental Advancements|
|Date:||Jan 1, 2013|
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