Disguised and Dangerous: A Case of Cervicofacial Actinomycosis Presenting With Hemoptysis

Abstract

Actinomycosis is a rare, chronic, suppurative bacterial infection caused by Actinomyces spp with an incidence of 0.3 cases per million people annually. The cervicofacial area is the most common site of infection and accounts for approximately 50 percent of all cases. Most cases of cervicofacial actinomycosis are odontogenic in origin.

Hemoptysis in the setting of actinomycosis commonly occurs from pulmonary involvement. Hemoptysis secondary to isolated cervicofacial actinomycosis has seldom been documented in literature. We hereby present a case of cervicofacial actinomycosis presenting with hemoptysis likely secondary to an oro-cervical fistula. We aim to highlight the challenges in diagnosis of this relatively rare clinical entity and the potential for life threatening complications if not detected and managed appropriately.

1. Introduction

Actinomycosis is a chronic, suppurative bacterial infection caused by Actinomyces spp with an incidence of 0.3 cases per million people annually. Actinomyces spp are constituents of normal oral flora and are increasingly prevalent in carious teeth and dental plaques.1 We hereby present a rare case of cervicofacial actinomycosis presenting with hemoptysis likely secondary to an Oro-cervical fistula. We discuss the challenges in diagnosis and the principles of management of this rare and life-threatening presentation.

2. Case presentation

A 59-year-old male with a known history of schizophrenia presented to the emergency department with a one-month history of a progressively enlarging, left-sided neck mass. The patient reported purulent drainage from the lesion for one week, accompanied by decreased oral intake and unintentional weight loss. He denied systemic symptoms, including fever, night sweats, cough, dyspnea, epistaxis, or otalgia. There was no history of recent travel or known sick contacts.

On examination, the patient was hemodynamically stable. Physical examination revealed a 5 × 5 cm necrotic, non-tender ulcerated lesion on the left side of the neck, surrounded by erythema and lacking local sensation. The intraoral evaluation demonstrated poor dentition with multiple carious teeth. Laboratory investigations (Table 1) were notable for leukocytosis, anemia, and metabolic acidosis. Lactic acid level was within normal limits.

Table 1.

Relevant patient investigations.

Investigation	Result	Normal range
WBC	17 × 109/L	(4 × 109/L–11 × 109/L)
Hemoglobin	8.2 g/dL	15 g/dL–18 g/dL
Serum pH	7.29	7.35–7.45
Serum Bicarbonate	17 mmol/L	21 mmol/L–28 mmol/L
Serum Lactate	1.0 mmol/L	0.5 mmol/L–1.6 mmol/L

Contrast-enhanced computed tomography (CT) of the neck (Fig. 1) demonstrated a complex, gas-containing fluid collection measuring 13 × 5.6 × 4.5 cm, extending from the skull base to the clavicle. CT imaging of the chest revealed multiple, bilateral, sub-centimeter (<3 mm), calcified pulmonary nodules. Given the imaging findings and infectious risk profile, airborne isolation was initiated due to concern for active tuberculosis. Subsequent HIV testing was negative. Empiric broad-spectrum antibiotics were started as well (see Figs. 2 and 3).

Fig. 1.

Contrast-enhanced computed tomography (CT) of the neck demonstrated a complex, gas-containing fluid collection measuring 13 × 5.6 × 4.5 cm, extending from the skull base to the clavicle.

Fig. 2.

A. The tissue shows basaloid groups of filamentous organisms (white arrows) in a background of necroinflammatory debris (black arrowhead) [H&E stain]. B. Grocott Methenamine Silver (GMS) stain lightly stains the organisms [white arrow].

Fig. 3.

T1-weighted MRI of head and neck demonstrating mandibular osteomyelitis (Black arrow).

Otolaryngology was consulted, and urgent surgical debridement of the cervical lesion was performed. Within 6 h postoperatively, the patient developed acute massive hemoptysis (~600 mL of clotted blood).

Conservative management was undertaken with tranexamic acid and a transfusion of two units of packed red blood cells; intubation was initially deferred as the patient declined the intervention. On hospital day 3, the patient experienced a second episode of hemoptysis with associated hemodynamic compromise, necessitating emergent intubation. He underwent tracheostomy and extraction of infected dentition. Intraoperatively, a fistulous tract between the floor of the mouth and the cervical wound was identified. Repeat debridement was performed and the lesion was closed over a penrose drain. Intraoperative tissue cultures grew mixed anaerobic flora and Propionibacterium acnes, though blood and respiratory cultures remained negative.

Histopathological analysis of debrided tissue from the ulcer and the left sternocleidomastoid muscle revealed filamentous bacteria with sulfur granules, consistent with cervicofacial actinomycosis. Antibiotic therapy was de-escalated to intravenous ampicillin-sulbactam. Sputum testing, including acid-fast staining and nucleic acid amplification testing (NAAT), was negative for Mycobacterium tuberculosis, leading to the discontinuation of airborne isolation.

Magnetic resonance imaging (MRI) of the head and neck revealed myositis of the masticatory muscles and osteomyelitis of the left mandible and temporal bone, including involvement of the temporomandibular joint (TMJ). A six-week course of intravenous antibiotics was recommended. On hospital day 11, the patient developed a non-pruritic maculopapular rash over the chest, suspected to be a hypersensitivity reaction to ampicillin-sulbactam, which was subsequently replaced with intravenous clindamycin and metronidazole.

Due to the extent of soft tissue damage, the Plastic Surgery team recommended delayed closure with a pectoralis major rotation flap following completion of antibiotic therapy. A percutaneous gastrostomy tube was placed by the interventional radiology team for continued nutrition. The patient was eventually discharged to a skilled nursing facility with a 6-week course of intravenous clindamycin. The patient was scheduled for outpatient follow-up with the infectious disease team to determine the appropriate duration of antibiotic therapy. Unfortunately, they were lost to follow-up after discharge.

3. Discussion

Cervicofacial actinomycosis, or “lumpy jaw,” accounts for approximately 50 % of actinomycosis cases and is most commonly caused by Actinomyces israelii.2 Infection is typically odontogenic in origin, with dental caries, gingivitis, or recent dental procedures acting as key risk factors. Immunocompromised conditions such as diabetes, malnutrition, alcohol use disorder may further predispose patients to infection. 3 In this case, poor dentition and multiple carious teeth likely contributed to disease development. Surgical disruption or instrumentation of tissue may also facilitate bacterial invasion, leading to chronic, indurated, suppurative infections with draining sinuses and fibrosis.4

Cervical masses present a broad differential diagnosis. While most (84 %) are benign, infectious etiologies account for approximately 37 % of these cases.5 Cervicofacial actinomycosis frequently involves the submandibular region and jaw angle, with potential extension to the masticator space, TMJ, and adjacent musculature.6 This correlates with our patient's findings of masticatory myositis, mandibular and temporal bone osteomyelitis, and necrosis of the sternocleidomastoid muscle.

Diagnosis is often delayed due to the non-specific and infiltrative nature of the disease, mimicking tuberculosis, nocardiosis, or malignancy. In our patient, negative acid-fast staining and cultures excluded mycobacterium and Nocardia infections. Additionally, the absence of lymphadenopathy supported actinomycosis, which typically does not respect fascial planes and spreads contiguously rather than via lymphatics.6 Histopathologic identification of sulfur granules—yellow, filamentous aggregates—is characteristic but not pathognomonic and may be seen in nocardiosis or botryomycosis.7

Although hemoptysis is common in pulmonary actinomycosis, our patient's calcified, sub-centimeter pulmonary nodules were radiographically inconsistent with pulmonary actinomycosis. Radiographic features of pulmonary actinomycosis can include segmental airspace consolidation with necrotic low-attenuation areas, adjacent pleural thickening & localized bronchiectasis with peribronchial consolidation. Multiple small cavities may also form within the lesion.6 Given the presence of a cervical-oral fistula identified intraoperatively, and the timing of hemoptysis following surgical debridement, it is likely that bleeding originated from the oropharyngeal cavity rather than the lungs. Additionally, it is unclear whether the hemoptysis is related to the initial debridement since both episodes of hemoptysis occurred only after the procedure. We advise caution while performing neck dissection in patients with a concern for fistulas. Hemoptysis as a potential complication must be kept in mind in these patients.

The first-line treatment for all forms of actinomycosis is intravenous penicillin G. In polymicrobial infections or when Actinomyces co-pathogens are suspected, ampicillin or amoxicillin-clavulanate is preferred. While treatment duration should be individualized, uncomplicated cervicofacial disease may respond to a shorter course of oral antibiotics (e.g., two months), potentially avoiding the need for surgical intervention.8,9

The compartmentalization of bacteria within granulation tissue and sulfur granules contribute to limited antibiotic penetration, often necessitating prolonged therapy.10 In our case, surgical intervention was required due to extensive tissue involvement, the formation of a cervical-oral fistula, and osteomyelitis. Close follow-up and adherence to a prolonged antibiotic regimen remain essential for ensuring resolution and optimizing reconstructive outcomes.

4. Conclusion

This case highlights the need to consider cervicofacial actinomycosis as a potential cause of an expanding neck mass, especially in the setting of poor oral hygiene and recent instrumentation. We also emphasize the need to rule out Oro-cervical fistulas given the severity of our patient's presentation. Prevention of this disease and its complications may require frequent evaluation of at-risk patients by a dentist and increased vigilance among clinicians managing these patients.1