Actinomycosis which Impersonates Malignancy.

Actinomycosis is a suppurative and often chronic bacterial infection most commonly caused by Actinomyces israelii. Actinomycotic infections may mimic more common oral diseases or present in a similar way to malignant disease. Treatment of actinomycosis involves surgical removal of the infected tissue and appropriate antibiotic therapy to eliminate the infection. Rarely seen in day-to-day dental practice, actinomycosis of the oral cavity is a highly significant condition due to its aggressive and locally destructive nature. We report a case of actinomycosis leading to extensive destruction and sequestration of the maxillary bone and deviation of the nasal septum, affecting a patient who complained of an unhealed extraction socket, chronic halitosis, and exposure of the bone with gingival recession crossing the midline.

INTRODUCTION

Actinomycosis of the maxilla and maxillary sinus is rare and usually arises from the oral cavity after dental procedures or trauma. More of all reported cases of actinomycosis involve the cervicofacial region. Human actinomycosis may pose a diagnostic problem at times and is often mistaken for a neoplasm. A case of fungal infection of the left maxillary sinus on the basis of history, clinical presentation, and radiologic findings has been reported. Tissue biopsy was negative for malignancy. Long-term penicillin treatment caused disappearance of all signs and symptoms. The report highlights the importance of bearing in mind the fact that certain rare, chronic, suppurative granulomatous infections, like actinomycosis, may mimic malignancy or a fungal infection.

CASE REPORT

A 70-year-old immunocompetent male patient was referred to our center for unhealed extraction socket, exposure of the left side of the maxillary alveolus, and chronic halitosis for 2–3 months. He had undergone uneventful extraction of teeth of the upper left posterior side of the jaw 3 months prior. However, in a private clinic, he was advised to take the medications and mouth wash for unhealed extraction socket and halitosis, and radiographs demonstrated erosion of the left maxillary bone. Halitosis persisted despite treatment with multiple antibiotics. The patient was afebrile. His maxilla was tender with exposure of bone crossing the midline from 15 to 26 due to necrosis [Figure 1]. There was no evidence of oro-antral fistula [Figure 2]. Nasal endoscopy revealed mucosal inflammation without pus in the region of the right middle meatus. Plain films of the sinuses showed left maxillary opacification, and a computed tomography scan was obtained [Figure 1]. The patient underwent a surgical excision of the exposed maxillary bone and an endoscopic exploration of nasal cavity [Figures 3 and 4]. The histology is shown in [Figure 5]. Actinomyces israelii was the only organism isolated. He was treated initially with antifungal due to fast necrosis of the bone till the histopathological examination reports revealed actinomycosis. Later, he was treated with a 3-week course of intravenous penicillin and further advised 1-month course of oral penicillin.

Figure 1

(a) Frontal view in the computed tomography scan shows necrosis of the right and left side maxillary bone involving the maxillary sinus (b) Side-view computed tomography scan reveals maxillary bone necrosis with no nasal bone involvement

Figure 2

Preoperative photograph showing receding gingiva on the buccal and the palatal aspect of the maxilla with unhealed extraction socket

Figure 3

Defect after surgical excision of the maxillary alveolar bone

Figure 4

Surgical excision of specimen

Figure 5

Histopathologic photomicrograph (H and E, ×25, for Actinomyces israelii)

DISCUSSION

Actinomyces are Gram-positive, predominantly anaerobic bacteria known to produce chronic suppurative infections in animals and human beings.[1] The term actinomycosis was derived from the Greek words aktino, because of the organism's radiating appearance in sulfur granules, and mykes, because it was mistaken for a fungal infection. Bollinger (1877) was credited with the discovery that Actinomyces bovis caused “lumpy jaw” in cattle. James Israel determined the causative agent in human beings that now bears his name, A. israelii. Other species infecting human beings, although less common, include Actinomyces odontolyticus, Actinomyces naeslundii, and Actinomyces viscosus. These organisms are not particularly virulent. Actinomycosis is characterized by the presence of sulfur granules within lesions, chronic granulomatous inflammation, and the presence of Actinomyces in culture. The organisms are usually seen as Gram-positive filaments after a Brown and Brenn Gram stain.[2] Actinomyces species are facultative intracellular bacteria that grow best in an anaerobic environment that distinguishes them from Nocardia which grow aerobically.[3] Actinomyces reside in the oral cavity of normal hosts and become pathogenic after a break of the mucosal barrier. Presumably, the infection spreads through direct extension to involve the paranasal sinuses.[4] The sinus mucosa becomes adherent to the underlying bone, which can become markedly firm. In this case actinomycosis probably resulted from the surgical trauma associated with the extraction of teeth. In the present case, the maxillary sinus was mostly involved, infections follow dental extractions. Infections in the patient received combination of surgical therapy and long-term antibiotics.

The diagnosis of craniofacial actinomycosis begins with a high degree of suspicion. Clinical features include a history of dental disease, facial swelling, and draining fistulae.[5] However, actinomycosis of the head and neck can resemble carcinoma, Wegener's granulomatosis, and congenital cysts.[6] Sinus involvement should be suspected when a patient with chronic sinusitis does not respond to medical therapy and has a history of dental disease or facial trauma. Nasal endoscopy should be performed, and direct cultures should be obtained if pus is found.[78] The cultures must be brought directly to the laboratory under anaerobic conditions and plated on brain–heart or blood agar. Radiographic findings are nonspecific but can be useful for surgical planning. The diagnosis is usually confirmed by the isolation of Actinomyces in culture. However, Actinomyces can be difficult to isolate, especially when synergistic organisms are present.[9] Occasionally, the diagnosis must be established on the basis of the presence of sulfur granules and the histologic findings after sinus surgery. Before the availability of antibiotics, many patients would die of their chronic infections. Radiation therapy and antral irrigations with various chemicals had limited success. In the past, penicillin changed the course of this disease and when used in combination with surgery, provided an effective means of treatment.[10] Surgical treatment has been surgical excision of the exposed alveolar maxillary bone as there was sinus involvement. After surgical therapy, intravenous penicillin is recommended for 2–6 weeks. Primary oral penicillin is then required for an additional 3–12 months, depending on the severity of the disease and treatment response. Tetracycline or clindamycin are excellent choices for the patient who is allergic to penicillin. Because Actinomyces grows best in an anaerobic environment, the endoscopic establishment of patent natural sinus ostia may contribute to treatment success. Also, office endoscopy may be used to monitor a patient's recovery and to detect recurrence. However, bone involvement is difficult to determine, and prolonged treatment with antibiotics is recommended.

CONCLUSION

A thorough case history, clinical examination, radiological findings coupled with biopsy should elude the diagnostic dilemma of actinomycosis that impersonates malignancy in head and neck region.

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Conflicts of interest

There are no conflicts of interest.