Case Report
Actinomycosis of the mandible
Naveen Sharma1, Seema Singh1, Devendra Kumar Ravi1, Mohan Kumar2, Manoj Pandey1
- 1Departments of Surgical Oncology, Institute of Medical Sciences, Banaras Hindu University, Varanasi 221 005, India
- 2Departments of Pathology, Institute of Medical Sciences, Banaras Hindu University, Varanasi 221 005, India
- Submitted: April 30, 2012;
- Accepted May 21, 2012
- Published: June 3, 2012
This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
Abstract
Introduction: Actinomycosis is a rare, slow, progressive disease that mimics neoplasia. Actinomycosis infections of the bone are even rarer and are seen mostly in immunocompromised patients.
Case presentation: A 50 year old male underwent total glossectomy with vein preserving neck dissection and pectoralis major myocutaneous flap reconstruction through a lateral mandibulotomy approach followed by external beam radiotherapy for T3N0M0 carcinoma of the anterior tongue. Two years later he developed an orocutaneous fistula with exposure of dead mandibular bone. The dead bone was excised and histopathology revealed it to be actinomycosis, patient was started on oral penicillin and is disease free at present.
Conclusion: Osteonecrosis of the mandible is a rare complication of radiotherapy and bisphosphonate therapy. Super infection with actinomycosis is rare. Removal of the dead bone and treatment with oral penicillin is the treatment of choice.
Introduction
Actinomycosis is a slowly progressive bacterial infection that usually manifests as a soft tissue swelling, abscess, or mass lesion that is often mistaken for a neoplasm. The most common site is cervicofacial region (55%) with a particular predilection for the angle of the jaw [1]. Actinomycosis was widely prevalent in the pre-antibiotic era but the incidence decreased drastically with the discovery of penicillin as the organisms were exquisitely sensitive to penicillin. The disease has shown resurgence in patients with HIV infection, transplantation, and patients receiving radio- or chemotherapy. There are more than 30 species of the organism; commonly seen species are A. israelii, A. naeslundii, A. odontolyticus, A. viscosus, A. meyeri, and A. gerencseriae [1]. We report a case of cervicofacial Actinomycosis following radiotherapy in a case of carcinoma tongue.
Case report
A 50-years-old gentleman presented with an ulceroproliferative lesion of the tongue, punch biopsy showed it to be squamous cell carcinoma, clinical stage cT3N0M0. He underwent total glossectomy with vein and nerve preserving neck dissection and pectoralis major myocutaneous flap reconstruction via lateral mandibulotomy and mandibular swing approach. The mandibular fracture was fixed with a titanium plate. Postoperative histopathology showed it to be well differentiated squamous cell carcinoma, pT3N0 with involved posterior margin. The patient received 60 Gy/30 Fr adjuvant external beam radiotherapy with cobalt. Following completion of treatment the patient was on regular follow-up.
Figure 1: Clinical photograph showing lesion with necrosed bone and the titanium plate that has been used for fixing mandibulectomy.
Figure 2: Photomicrograph showing actinomycosis of the bone. (Hematoxylin and Eosin X40).
Two years later he developed a gradually progressive intra oral ulcer in the region of the floor of the mouth, a recurrence was suspected and a punch biopsy was taken that showed chronic inflammatory changes, no viable tumor was present. In view of strong clinical suspicion and appearance of lesion biopsy was repeated which again showed a similar picture. He was started on chemotherapy with cisplatin and 5 FU despite a negative biopsy. After 6 cycles of chemotherapy the lesion showed no regression or progression, a repeat biopsy was again inconclusive. In view of this it was decided to stop the chemotherapy, and put him on close follow-up. Three months later, he presented with another lesion on the skin above the right angle of the mandible with pus discharge and an oro-cutaneous fistula (figure 1) with exposure of mandibular plate and dead necrosed bone. He was clinically diagnosed as having osteoradionecrosis of the mandible. He underwent excision of the necrosed part of the mandible with removal of the plate. The histology report of the excised necrotic mandible came out to be Actinomycosis of the mandible (Figure 2). The patient was treated with oral penicillin and the fistula healed spontaneously. The patient is disease and symptom free three years after the diagnosis of actinomycosis and 5 years after his initial surgery for carcinoma of the tongue.
Discussion
Osteonecrosis of the facial bones is usually seen in the setting of cervico facial radiotherapy. Rarely maxilla-mandibular osteonecrosis can be seen in association with bisphosphonate use [2]. Actinomycosis is considered a disease of the past with improved dental hygiene and early referral however; there has been resurgence in its incidence in immunocompromised patients. The lesion is characterized by presence of sulphur granules that are demonstrated on microscopy and sometimes clinically. Actinomyces are a normal commensal of the oral cavity, female genital tract and lower gastrointestinal tract. Whenever there is a breach in the mucosa, the bacteria may become invasive, more so in the immunocompromised patients [3, 4]. The exact pathogenesis of invasive actinomycosis is not known [5]. The treatment of Actinomycosis involves long term antibiotic therapy, usually penicillins, but the rate of healing of osteoradionecrosis in the presence of actinomycosis is lower [6]. Actinomycosis closely resembles osteoradionecrosis clinically except for the presence of sulphur granules [7]. It is well established that irradiated patients are high-risk for Actinomyces infection, one study correlated osteoradionecrosis and actinomycosis [8]. Clinically, histologically and radiologically, Actinomycosis can resemble tumor unless specifically suspected and looked for it [9].
Our patient was a high-risk for developing Actinomycosis because of post surgery, post radiotherapy status, but his disease manifested almost two years after completion of therapy.
Conclusions
Actinomycosis should be considered as a differential diagnosis in cases of osteomyelitis of the jaw. A high index of suspicion is essential to make a diagnosis and start treatment.
Authors’ Contributions
NS: Prepared the draft manuscript
SS: Literature review and preparing of manuscript
DKR: Literature review and preparing the draft manuscript
MK: Pathology of the resected specimen and pathology part of manuscript, editing of manuscript
MP: Conceived and designed the study and final editing and revision of manuscript.
Conflict of Interests
The authors declare that there are no conflicts of interests.
Ethical considerations
Written informed consent was obtained from the patient for publication of this case report and photographs.
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