Cervicofacial actinomycosis is a chronic, suppurative, granulomatous infection caused by Gram-positive bacteria named Actinomyces israelii, which is an inhabitant of normal oral flora. 1–3 The lesion is characterized by draining sinuses, fibrosis, and pus-filled abscesses that often contain characteristic multilobulated “sulfur” granules. The clinical picture of actinomycosis can resemble a variety of pathologies, ranging from malignant or benign tumors to various granulomatous or mycotic infections. 4 The common use of penicillin and other antibiotics for various infections of the head and neck may have reduced the prevalence and changed the clinical course of an actinomycotic infection. We present two cases of cervicofacial actinomycosis and emphasize diagnostic and therapeutic problems.
A 32-year-old white man was admitted to our clinic with a mass at the left frontal region that continued to enlarge over several weeks after removal of a lesion at the same site without any diagnosis. The lesion had invaded the upper part of the left upper lid and the lower part of the left frontal region. The mass was hard, reddish yellow with irregular contours, and demonstrated multiple sinuses with partial intact skin over the swelling. The lesion was slightly tender and ill defined, measuring approximately 5 × 6 cm (Figs A, B).There was no area of fluctuation and no fluid could be obtained from the swelling. The patient’s history revealed blunt trauma to his left forehead before the appearance of any wound. Clinical findings suggested a malignant tumor. A biopsy was performed, and a diagnosis of actinomycosis was confirmed by histopathological examination (Fig D).The patient was started on intravenous penicillin G (12,000,000 U per day), and the lesion was excised completely (Fig E). The defect was reconstructed by rotating a frontogaleal flap, which was then covered with a split-thickness skin graft. The patient was continued on intravenous penicillin G for 10 days followed by oral penicillin V (4 g per day) for two more weeks. Two months later, the patient was admitted to the hospital with a recurrence of the mass, which demonstrated suppuration, intense cicatrization, and thickening of the soft tissue (Fig F). Reexcision of the lesion was performed down to the frontal bone. The defect was reconstructed with a free radial forearm flap (Fig G). The patient was again started on intravenous penicillin G (12,000,000 U per day) for 2 weeks, and continued with oral penicillin V (4g per day) for 2 months. The lesion has not recurred for more than 4 years.
A 36-year-old white woman was referred to our clinic because of a progressive swelling at the left parotid region that had started approximately 4 weeks earlier. The lesion showed diffuse swelling with induration. There was slight fluctuation of the mass with ill-defined margins. There was no discoloration or trismus, and her facial nerve function was normal. There was no inflammation of the left Stensen’s duct, and intraoral examination revealed no pathology. Her orthopantomograph was entirely normal, without any evidence of fracture, osteomyelitis, or apical abscess. She did not have a history of recent tooth extraction or trauma to her face. Three days later, when she was admitted to the hospital, there was more facial swelling on the left side, with obvious fluctuation and discoloration. The mass was drained of a yellowish brown serosanguinous exudate. The mass was still firm and indurated by palpation (Fig C). The pus draining from the lesion was taken for microscopic examination. Gram-positive mycelial filaments characteristic of A. israelii were identified. The patient was started on intravenous penicillin G (12,000,000 U per day), which continued for 10 days. The mass started to soften and decrease in size. She was discharged with oral penicillin V (4 g per day), and this therapy continued for 2 months, at which time there was complete resolution. Her 3-year follow-up did not reveal any recurrence.
Cervicofacial actinomycosis still occurs occasionally and the lesion can mimic many other diseases, including neoplasms of the head and neck. Thus, diagnosis may be difficult.
Actinomycotic infection is usually caused by tooth extraction, odontogenic infection, or trauma. 2 The etiology was unclear in our patients because they had no identifiable portal of entry. Most authors classify actinomycosis according to the anatomic location of the lesion in cervicofacial, abdominal, and pulmonary forms. 3,5 Forty percent to 60% of actinomycotic infections occur in the craniofacial skeleton, 6,7 and the mandible is the site of predilection. 3,8 The literature reveals that although actinomycosis may be seen in the parotid and masticator spaces, 9–11 a few cases of the lesion have also been reported in the temporal region, 12 paranasal sinuses, 13 palate, 14 and tongue. 15 However, occurrence of actinomycosis primarily in the frontal region, as in our Patient 1, has not been reported previously. Actinomycosis in this region is important because the lesion may spread over the years and involve cranial bone. 2,12
Two patterns of cervicofacial actinomycosis are defined clinically. One is chronic, slowly progressive, and indurated with multiple abscesses and fistulas (“typical”), as in Patient 1, and the other is the more acute, rapidly progressive type with a fluctuating mass, as in Patient 2. 16 It may be speculated that Patient 2 presented an acute exacerbation of chronic actinomycosis with low virulence. Acute cervicofacial actinomycosis seems to be treated more readily than chronic cervicofacial actinomycosis—a finding also reported by others. 5,16 Widespread use of antibiotics has caused considerable changes in the clinical course of this lesion, and these 2 patients demonstrate the variability of the clinical manifestation and location.
Direct involvement of major salivary glands by actinomycosis is rare. Fenton and Rotenberg 9 believe that some of the cases of actinomycosis of the parotid region are in fact infections involving the masticator space, but this is difficult to distinguish clinically.
Actinomycosis is diagnosed by examining the exudate and infected tissue. “Sulfur granules” are characteristic. 1 False-negative results may be caused by the wrong procedure of sampling and/or transporting specimens. 17 Intensive, prolonged antibiotic therapy and drainage or surgical removal of the lesion are the basic forms of therapy. The antibiotic of choice is penicillin, but clindamycin, erythromycin, and tetracycline may also be used. 2,16 Duration of antibiotic therapy depends on the patient’s general condition and on the remission of pathological changes. Antibiotics may be prescribed for 2 to 3 months, as recommended in the literature. 6,17,18
The outermost periphery of the lesion can be intensely fibrotic, collagenous, and remarkably avascular. It is this sclerosis and poor vascularity that act as a harbor for the infecting organism, and prevent the penetration of antibiotics and keep the lesion anaerobic. 2,6 This explains why antimicrobial therapy may fail and the lesion may recur, as it did in Patient 1. Some authors have recommended the use of hyperbaric oxygen. 3,19
In conclusion, the patients presented here emphasize the difficulty in the diagnosis and therapy of actinomycotic lesions. Actinomycosis should be considered in the differential diagnosis for every unidentified mass or swelling in the cervicofacial region. We think that the choice of treatment is radical resection of the lesion in chronic cases and drainage in acute cases. Penicillin remains the antibiotic of choice, but antibiotic therapy must be followed strictly by the patient.
1. Regezi JA, Sciubba J. Oral pathology. 2nd ed. Philadelphia: WB Saunders, 1993: 45–46
2. Miller M, Haddad AJ. Cervicofacial actinomycosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1998; 85: 496–508
3. Nagler R, Peled M, Laufer D. Cervicofacial actinomycosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997; 83: 652–656
4. Deloah-Banta LJ, Barber FA. Nonhealing masses of the right cheek and submandibular areas. Arch Dermatol 1991; 127: 1831–1834
5. Norman JE. Cervicofacial actinomycosis. Oral Surg 1970; 29: 735–745
6. Bennhoff DF. Actinomycosis: diagnostic and therapeutic considerations and review of 32 cases. Laryngoscope 1984; 94: 1198–1217
7. Chuong R, Goldberg M. Case 60: preauricular mass (clinicopathological conferences). J Oral Maxillofac Surg 1986; 44: 214–217
8. Holst E, Lund P. Cervico-facial actinomycosis: a retrospective study. Int J Oral Surg 1979; 8: 194–198
9. Fenton RS, Rotenberg D. Actinomycosis of the parotid. J Otolaryngol 1977; 6: 233–238
10. Sazama L, Kralove H. Actinomycosis of the parotid gland; report of five cases. Oral Surg 1965; 19: 197–204
11. Hopkins R. Primary actinomycosis of the parotid gland. Br J Oral Surg 1973; 11: 131–138
12. Zajc I, Orihovac Z, Bagatin M. Temporal actinomycosis: report of a case. J Oral Maxillofac Surg 1999; 57: 1370–1372
13. Roth M, Montone KT. Actinomycosis of the paranasal sinuses: a case report and review. Otolaryngol Head Neck Surg 1996; 114: 818–821
14. Herman WW, Whitaker SB, Williams MF, Sangueza OP. Acute actinomycosis presenting as an ulcerated palatal mass. J Oral Maxillofac Surg 1998; 56: 1098–1101
15. Ficarra G, DiLollo S, Pierleoni F, Panzoni E. Actinomycosis of the tongue: a diagnostic challenge. Head Neck 1993; 15: 53–55
16. Nielsen PM, Novak A. Acute cervico-facial actinomycosis. Int J Oral Maxillofac Surg 1987; 16: 440–444
17. Bartkowski SB, Zapala J, Heczko P, Szuta M. Actinomycotic osteomyelitis of the mandible: review of 15 cases. J Craniomaxillofac Surg 1998; 26: 63–67
18. Hartley JH, Schatten WE. Cervicofacial actinomycosis. Plast Reconstr Surg 1973; 1: 44–50
19. Kawai M, Mizutani H, Ueda M, et al Cervicofacial actinomycosis: report of two cases. Nagoya J Med Sci 1993; 55: 83–88