Unusual Behavior of a Posttraumatic Scar: Craniofacial Actinomycosis

Actinomycosis is a chronic suppurative granulomatous infection most commonly involving the cervicofacial region. Clinical diagnosis is usually difficult, and fine-needle aspiration cytology or imaging studies are usually unhelpful in diagnosing actinomycosis. Definitive diagnosis is based on the histopathological examination of a tissue biopsy. The authors report a case of a 32-year-old healthy man who underwent multiple surgeries over a period of 7 years to correct a posttraumatic scar on his forehead with unusual behavior. Final diagnosis was made by tissue biopsy. Scar was excised and penicillin was administered for 1 month postoperatively; after a 12-month follow-up, the wound was fully healed with minimal scarring and no recurrence.

Scar formation is an inevitable consequence of wound healing. Although desired by both the patient and surgeon, an unnoticeable scar is not always achievable due to certain interferences in wound healing that lead to disfiguring scars such as ischemia, infection, foreign bodies, steroids, diabetes, smoking, etc. After excluding these aforementioned conditions and ensuring optimal surgical technique, the surgeon must consider other diagnoses if the scar does not mature naturally. The authors report a case of a 32-year-old healthy man who underwent multiple surgeries over a 7-year period to correct a scar from a road traffic accident over his forehead with multiple discharging sinuses, which lowered the patient’s self-esteem. Definitive diagnosis of actinomycosis was made by tissue biopsy after ruling out medical and hereditary conditions. He required both surgical and medical treatments for actinomycosis, and after a 12-month postoperative follow-up, the wound was well settled with no recurrence, thereby alleviating the patient’s poor self-esteem due to his scar.

A 32-year-old healthy man presented as an outpatient to the authors’ Department of Plastic Surgery with a 10 cm x 6 cm scar over the right side of his forehead. The scar extended to the right upper eyelid, with multiple palpable nodules and intermittent seropurulent discharging sinuses that had lasted for the past 3 years. The scar was tender and fixed to underlying bone with surrounding induration and inflammation. The patient had suffered a road traffic accident 7 years prior in 2008, and the wounds healed by secondary intention, which led to hypertrophic scarring. He was asymptomatic for 2 years before noticing a painless nodular swelling over the scar on the right side of his forehead in 2010; excision of the swelling and scar revision were performed. Histopathological studies revealed chronic granulomatous tissue. The patient again had recurrence of swelling in December 2011, and investigations ruled out tuberculosis and other comorbid conditions. Repeat biopsy was also reported as granulomatous tissue. In February 2012, he underwent fine-needle aspiration cytology (FNAC) of the swelling for persistent symptoms, which was inconclusive. Intralesional triamcinolone (40 mg) injection was administered, and, following this, the patient gradually developed a fluctuant swelling in the scar, which was drained and allowed to heal secondarily. The patient had recurrence of discharge from the same site after 1 year in 2013, and the size of the swelling gradually increased with multiple discharging sinuses (Figure 1). 

At this point, the patient presented to the authors’ institution and underwent surgical excision of the scar tissue. Inraoperatively, deep multiple sinuses were found extending to the periosteum without bony involvement. Histopathological examination revealed chronic inflammation with the presence of multiple granules surrounded by polymorphocytes; microscopic findings were consistent with diagnosis of actinomycosis. Postoperatively, the authors started the patient on intravenous penicillin G at 10 million U/d divided and administered every 6 hours for 4 weeks. He responded well to the treatment and had an uneventful recovery. No recurrence has developed, and the 12-month follow-up revealed a healthy scar.

Actinomycosis is an endogenous infection caused by gram-positive, nonacid fast, anaerobic, or microaerophilic filamentous-branched bacteria of low pathogenicity.¹ The most common pathogen is Actinomyces israelii, followed by Arachniapropionica, A naeslundii, A viscosus, and A odontolyticus that are less common. These bacteria are normal commensals of the human oral cavity. Actinomycosis is seen more often in males and young adults. No person-to-person transmission, racial predisposition, or geographic factors have yet to be documented.² Malnutrition, radiation therapy, alcoholism, and debilitating states such as diabetes, malignancy, and immunosuppression are predisposing factors. Tissue injury is a prerequisite for the infection and hence more common after maxillofacial trauma, surgical procedures, and dental manipulations.³

Cervicofacial actinomycosis is the most common form and sites frequently involved are submandibular space, cheek, parotid gland, teeth, oral cavity, and nasal cavity.⁴ Symptoms are often nonspecific, with mild pain and low-grade fever seen in > 50% of patients.⁵ It may be associated with a sensation of superficial tension around the mass and become tender at a later stage due to central necrosis.⁶

The diagnosis of actinomycosis is usually difficult. The classic formation of spontaneous sinus tracts draining purulent material is observed in only 40% of cases and may be helpful in differential diagnosis.⁷ Actinomycosis can present both clinically and radiologically in a variety of forms and may mimic other infections such as tuberculosis, chronic granulomatous lesions, fungal infections, and even malignancy.⁸ Moreover, it is very difficult to grow Actinomyces in culture, with < 30% of cultures being positive.⁹ Imaging techniques (computed tomography scan and magnetic resonance imaging) usually yield nonspecific findings, only contributing to defining the radiological features of the mass, its extension, and its involvement in adjacent soft tissues, though the techniques help plan the treatment as in this reported case.⁷

The main stay of diagnosis continues to be histopathological examination of the biopsy specimen, especially in complex cases like the present one with noncontributory history, previous investigations, and FNAC. A central neutrophilic lobulated abscess with a number of sulfur granules surrounded by granulation tissue is seen. Actinomyces sulfur granules appear as basophilic structures with elongated eosinophilic clubs, which radiate from the periphery, on sectioned stained with hematoxylin and eosin.¹⁰ Due to these numerous pitfalls in the diagnosis, cervicofacial actinomycosis has been referred to as the great trickster of head and neck diseases because < 10% of infections have been correctly diagnosed.⁷

Penicillin is the primary drug for treating actinomycosis,¹¹ though tetracycline and erythromycin can be employed in patients allergic to penicillin. In the acute phase, penicillin can be replaced by third-generation cephalosporins, which are effective in the presence of coinfections with other bacteria. Surgical excision remains the definitive treatment,¹² particularly in cases presenting with the formation of an abscess after steroid injection, unresponsive to antimicrobial therapy, or when FNAC is nondiagnostic. In the authors’ case, the leasion was surgically excised for definitive diagnosis, and complete resolution of symptoms was achieved after adequate postoperative antibiotic treatment (Figure 2). 

At present, actinomycosis is considered a rare disease and mimics numerous common conditions in human pathology. When it is not an occupational disease (agriculturists, butchers, breeders), diagnosis is difficult. Symptoms, clinical examination, radiological imaging, and FNAC are less helpful, necessitating a tissue biopsy for confirmation of diagnosis. Treatment requires surgical excision, penicillin, and supportive measures, as done in this case. The authors conclude that actinomycosis should be considered in all nonhealing or abnormally behaving scars.

From the Division of Plastic Surgery, Sir Gangaram Hospital, New Delhi, India 

Address correspondence to:
Ritesh Panda, DNB
DNB Plastic Surgery
Sir Ganga Ram Hospital
Room No. 2325
Old Rajender Nagar
New Delhi 110060, India
dr.riteshpanda@gmail.com
ritesh.panda@yahoo.com

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