Actinomycosis is a chronic granulomatous infection that commonly occurs in the cervicofacial region. Although Actinomcyes is an element of the normal oral flora, infections of the facial skin are very rare because of the entirely endogenous habitation of the organism. The authors report a case of facial actinomycosis, which mimicked a cutaneous tumor both clinically and surgically in a 44-year-old woman with chronic renal failure and Hepatitis C viral infection.The majority of cases can be treated with long-term antibiotics. However, a treatment-resistant abscess, a fistula, or postsurgical excision of the mass formation that are infected can be treated with antibiotics as soon as possible, and recurrence of infection is prevented. The treatment should consist of conservative surgery to obtain a firm histological diagnosis and to drain any collections.

Actinomycosis is a chronic granulomatous infection that commonly occurs in the cervicofacial region.¹ It can also occur in abdominopelvic and pulmonothoracic sites and in the musculoskeletal and central nervous system. Any part of the cervicofacial site can be affected by actinomycosis infection.  Although Actinomyces is an element of normal oral flora, infections of the facial skin are very rare because of the entirely endogenous habitation of the organism. However, in some cases actinomycosis can be locally destructive and progress to generalized infections, causing complications including abscess formation with risk of rupture, fistula formation, or spread to neighboring and distant organs.² The most frequently encountered species is Actinomyces israelii.^3,4 

The authors describe an unusual case of a 44-year-old woman with chronic renal failure and Hepatitis C viral (HCV) infection having primary cutaneous actinomycosis (PCA) of the left cheek. Infection probably occurred following direct inoculation of the bacteria after a previous surgery. The patient was diagnosed and successfully treated with surgical excision followed by antibiotic treatment.

A 44-year-old woman with chronic renal failure and HCV infection presented to the authors’ outpatient clinic of the Plastic Surgery Department with a history of a mass in the left cheek for 16 months. She had an operation due to a similar complaint in the same location 3 years prior, but a pathology report was not recorded. Physical exam revealed a 2-cm, nontender, soft cheek mass (Figure 1). The remainder of the head and neck exam was normal. Any extracutaneous lesions and lymphadenopathy were not detected. The patient had no history of diabetes mellitus, dental surgery, trauma, or tuberculosis. All microbiology tests were negative; serological and hematological investigations were normal. The patient was then taken for open biopsy of the mass. Histopathological examination of the tissue sample revealed acute necrotizing and chronic inflammation. The presence of aggregates of filamentous basophilic microorganisms in “sunburst fashion” were suggestive of actinomycosis (Figure 1). The patient consulted with an infectious disease clinic, and there were no signs of malignancy. The lesion did not significantly change despite receiving high doses of penicillin treatment (crystalline pen, 20 mIU/day) for 1 month; after penicillin treatment was complete, clindamycin (1800 mg/day) was administered for 14 days. The lesion was excised surgically, and the defective area was closed with a skin flap (Figure 2). After 30 days of medical treatment with the same drugs, the patient remained in good health, and there was no recurrence after 5 years of follow-up. 

Primary cutaneous actinomycosis is very rare, and risk factors include dental surgery, immunosuppression, maxillofacial trauma, local ischemia, and the presence of a foreign body.⁵ Actinomycosis occurs following damage to the skin or mucosa, leading to abscess formation with draining sinus tracts, fistulae, or mass lesions that are often misdiagnosed as cutaneous tumors.^5-8 However, the patient’s prior facial surgery and her immunosuppression from chronic renal failure were the determining elements to suggest the actinomycosis diagnosis.^4,9

Although actinomycosis is not actually an “opportunistic” disease, many reports^3,10-12 of actinomycosis in immunosuppressed patients have been published recently. Actinomycosis without a history of surgery or trauma may indicate the need to evaluate an underlying immunological deficit. Early diagnosis remains important for extensive surgical treatment and decreased morbidity. If workups for tuberculosis and fungal infections are negative, a high degree of suspicion is needed to diagnose actinomycosis correctly. 

Actinomycosis frequently imitates a neoplastic process when faced with an ill-defined and fixed mass. Facial forms are difficult to diagnose since the clinical presentation is often nonspecific and highly variable, but also because of a general lack of experience with the disease and the complicated microbiological identification.² Although the definitive diagnosis of actinomycosis comes from positive cultures, a negative culture result does not exclude the diagnosis,¹³ as in this case where the culture result was negative. The diagnosis is most often made by the histological examination of tissue samples since the presence of characteristic clusters of organisms with sulfur granules is pathognomonic.¹⁴ Sulfur granules with Actinomyces colonies were shown in the histological examination in the case herein.  

The treatment of a classical case of cutaneous actinomycosis includes high doses of intravenous antibiotics for 4–6 weeks followed by oral penicillin or amoxicillin for 6–12 months. In penicillin-resistant cases, the other alternatives are clindamycin, tetracycline, erythromycin, and chloramphenicol.^15-17 The present patient received high doses of penicillin for 1 month and then clindamycin for 14 days. In the majority of the cases, surgical excision may ultimately be required for definitive diagnosis and treatment of actinomycosis. The prognosis depends on quick treatment. The infection is highly responsive to antibiotics, including penicillin and clindamycin, but prolonged treatment is needed in most cases. Surgical treatment is certainly necessary; however, cases complicated by fistula or abscess formation need to debulk necrotic and fibrotic tissues unresponsive to antibiotic treatment or to diagnose.² Surgery alone is not curative.¹⁸ For this reason, there is a need for the combination of surgical intervention and antibiotics. With this dual treatment, better cosmetic results are obtained and the rate of recurrence is reduced.^17,18

In patients presenting with a facial cutaneous mass, the initial diagnosis of a skin tumor is highly possible. Therefore, a more extensive surgical resection of such mass and complex surgical reconstructions are inevitable. To avoid misdiagnosis and treatment, other pathologies are needed. Consequently, the majority of cases can be treated with long-term antibiotics. However, resistant abscesses, fistulae, or infected postsurgical mass excisions should be treated with antibiotics as soon as possible so recurrence of infection is prevented. The treatment should consist of conservative surgery to obtain a firm histological diagnosis and to drain any collections.

From the Pamukkale University Hospital, Department of Plastic, Reconstructive and Aesthetic Surgery, Denizli, Turkey

Address correspondence to:
Adem Özkan, MD
Pamukkale University Hospital
Department of Plastic, Reconstructive and Aesthetic Surgery
20070 Pamukkale, Denizli, Turkey
drademozkan@gmail.com

Disclosure: The authors disclose no financial or other conflicts of interest.