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Actinomycosis of the pharynx.

Abstract

Few cases of pharyngeal actinomycosis have been documented in the literature. We describe the case of a 67-year-old white man who presented with symptoms of dysphagia. Laryngoscopy revealed a pedunculated mass in the left posterior pharyngeal wall; an excisional biopsy confirmed the diagnosis. Postoperatively, the patient underwent 10 weeks of intravenous penicillin therapy followed by 4 months of oral antibiotics, and his condition resolved. We discuss the diagnosis, management, and complications of this rare infection.

Introduction

Actinomyces, a gram-positive bacterium, rarely causes infections. Actinomycosis appears to have no geographic or racial predilection, but there is a 3:1 predilection for males. (1,2) Cervicofacial actinomycosis is the most common form of this disease, with most of these cases occurring in the parotid gland, cheek, and submandibular space. (1,3,4) In this report, we present a new case of Actinomyces infection of the posterior pharynx.

Case report

A 67-year-old white man presented to our ENT clinic complaining of gradually progressive dysphagia for solids and liquids and a foreign-body sensation. He had recently worked in Haiti for 3 months helping with an earthquake relief effort. He believed that his symptoms were related to his work there, which involved extracting bodies and clearing rubble. He denied odynophagia, cough, hemoptysis, voice changes, fever, and chills. His medical history was unremarkable; he drank 4 alcoholic beverages per week and he had never smoked.

Upon examination, the patient had a healthy appearance. He was breathing comfortably without stridor, he was in no acute distress, and he was afebrile. His neck was supple, with no lymphadenopathy or masses and no skin erythema. His oral cavity and oropharynx were clear.

Laryngoscopy demonstrated a pedunculated mass in the left posterior pharyngeal wall that partially obstructed the airway (figure). An awake biopsy was performed with flexible forceps. The biopsy identified numerous bacterial colonies consistent with actinomycosis. Neither fungal organisms nor malignancy was observed on periodic acid-Schiff (PAS) and Gomori methenamine silver (GMS) staining.

Because the patient's symptoms continued to worsen, he was brought to the operating room the following day for an excisional biopsy, which confirmed the original findings. Postoperatively, he was given a peripherally inserted central catheter (PICC line) and started on intravenous penicillin G at 12 million U/day by continuous infusion for 5 weeks. He was discharged on postoperative day 1.

When the 5 weeks were up, the patient returned to the hospital complaining of increasing throat irritation, choking, and dysphagia. His PICC line was still in place, and he had not missed any treatment sessions since his discharge. A repeat laryngoscopy revealed evidence of a recurrence, as a pedunculated mass on the posterior pharyngeal wall had prolapsed into the posterior larynx. A second surgical excision was performed, and the patient's penicillin regimen was increased to 24 million U/day by continuous infusion for another 5 weeks. Following the second course of intravenous therapy, the patient was prescribed 4 months of oral antibiotic therapy. At the end of treatment, his symptoms had completely resolved.

Discussion

For many years, Actinomyces organisms were incorrectly believed to be fungi; indeed, in Greek, Actinomyces translates to ray fungus. In fact, these organisms are anaerobic, non-acid-fast, filamentous, branching bacteria. (2)

The order Actinomycetales includes the families Mycobacteriaceae and Nocardiaceae, among others. The organisms in these two families in many ways mimic Actinomyces. For example, like Actinomyces organisms, Nocardia organisms can form sulfur granules. However, these two types can be differentiated by their acid-fast and aerobic properties. (4)

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In humans, the four main clinical types of actinomycosis are cervicofacial, thoracic, abdominopelvic, and central nervous system (CNS). The cervicofacial type is the most common, accounting for about 55% of all cases. (4) These bacteria naturally exist in high concentrations in the tonsillar crypts and gingivodental crevices. They cannot penetrate healthy tissue, and therefore disease occurs only when a mucosal injury allows them to invade the subcutaneous tissue. Such an injury introduces these organisms to an anaerobic environment where they can thrive.

Risk factors for cervicofacial actinomycosis include poor dental hygiene, dental procedures, chronic tonsillitis, otitis, mastoiditis, maxillofacial trauma, and immunocompromise. (3,4) No case of person-to-person transmission has been recorded in the literature. (2)

Actinomycosis specific to the pharyngeal area presents with symptoms of dysphagia, odynophagia, stridor, dyspnea, and hoarseness. A chronic form of the infection is most common, with slowly progressing and fluctuating symptoms that develop over weeks, months, or even years. (5) The infection may present with our without lymphadenopathy. Our review of the literature found that the infection rarely involves the lymph nodes because the large size of these organisms is not conducive to lymphatic spread. Instead, these organisms usually spread by direct extension. (3,6-9)

The differential diagnosis of actinomycosis includes fungal infections, neoplasms, Nocardia infection, and Mycobacterium infection, among others. (6) Because of the broad differential diagnosis and the deceptive nature of the organism, fewer than 10% of cases are diagnosed at the initial presentation. (8) This prompted some researchers to call actinomycosis "the great masquerader." (7)

A diagnosis can be made by culture and histologic examination of tissue. Cultures must be meticulously performed in a favorable anaerobic environment, but even then recovery rates reach only 30%. (8) Microscopic examination of biopsied tissue will show an outer zone of granulation and a central zone of necrosis. (7) Staining with PAS, GMS, and methylene blue will reveal long, branching filaments. (5) In addition, the characteristic sulfur granules are often seen on PAS and hematoxylin and eosin staining. (9,10) Sulfur granules, however, are not pathognomonic for Actinomyces because other organisms such as Nocardia, Sporotrichum, and Phialophora spp also produce them. (5)

Imaging studies may show the site and size of an actinomycotic infection. Park et al reviewed computed tomography (CT) and magnetic resonance imaging (MRI) scans in 7 patients with cervicofacial actinomycosis. (3) The CTs showed (1) ill-defined margins, (2) the organism's propensity for invading adjacent tissue places, and (3) relatively homogeneous contrast enhancement; the CTs also allowed for analysis of adjacent lymph nodes. The MRIs revealed moderate contrast enhancement. Enhancement may suggest areas of inflammation. Areas of low density, especially when centrally located, may indicate a necrotic center or an abscess. (11) In general, imaging demonstrates a nonspecific inflammatory soft-tissue mass.

Both medical and surgical therapies are available as treatment options. Before the advent of antibiotics, actinomycosis was much more prevalent than it is now. Today penicillin, which has been regularly used to treat actinomycosis since the 1940s, is the therapeutic gold standard. (5) It is routinely administered intravenously for several weeks, after which time the patient is switched to an oral dose. For years, the traditional duration of therapy was 1 year. However, more recent studies have demonstrated that shorter courses of therapy are just as effective in eradicating the disease. (12)

The shorter course has been found to be particularly successful in treating the cervicofacial form of the disease (92% eradication rate). (13) For patients who do not tolerate penicillin, the literature shows that tetracyclines are the preferred alternative, followed by erythromycin, cephalosporins, and imipenem. (8,13) The route and duration of treatment should be adjusted for each individual patient according to the course of the disease, recurrences, and clinical response to therapy.

Surgery plays a double role by providing a definitive diagnosis for the enigmatic organism, as well as by acting as a crucial aspect of treatment. However, surgery as a single mode of therapy has been associated with high rates of recurrence, so medical treatment usually accompanies surgical excision. (7) The surgical option should be considered when (1) the diagnosis is uncertain, (2) a neoplasm is suspected, (3) a fistula is present, (4) necrotic tissues need to be debrided, or (5) the patient is unresponsive to medical treatment. (1,2)

Since our patients lesion manifested in an easily accessible location in the pharynx that was conducive to an uncomplicated surgical excision, we opted for surgery in order to obtain immediate diagnostic and therapeutic relief, and we subsequently prescribed antibiotic therapy.

Complications of cervicofacial actinomycosis are rare; when they have occurred, they were life-threatening in some isolated instances. An infection can spread hematogenously or by direct extension from the primary site to the CNS. Actinomycosis of the CNS presents as a brain abscess in 75% of cases, but it can also manifest as meningitis or an empyema. (4) A cervicofacial or pharyngeal infection can also spread down the bronchus, invade the thoracic cavity, and involve the lungs, pleura, chest wall, and mediastinum. (14)

References

(1.) Lancella A, Abbate G, Foscolo AM, Dosdegani R. Two unusual presentations of cervicofacial actinomycosis and review of the literature. Acta Otorhinolaryngol Ital 2008;28(2):89-93.

(2.) Brook I. Actinomycosis: Diagnosis and management. South Med I 2008; 101 (10): 1019-23.

(3.) Park JK, Lee HK, Ha HK, et al. Cervicofacial actinomycosis: CT and MR imaging findings in seven patients. AJNR Am J Neuroradiol 2003;24(3):331-5.

(4.) Smego RA Jr., Foglia G. Actinomycosis. Clin Infect Dis 1998;26(6): 1255-61; quiz 1262-3.

(5.) Goldberg MH. Diagnosis and treatment of cervicofacial actinomycosis. Oral Maxillofac Surg Clin North Am 2003;15(1):51-8.

(6.) Everts EC. Cervicofacial actinomycosis. Arch Otolaryngol 1970;92 (5):468-74.

(7.) Volante M, Contucci AM, Fantoni M, et al. Cervicofacial actinomycosis: Still a difficult differential diagnosis. Acta Otorhinolaryngol Ital 2005;25(2):116-19.

(8.) Belmont MJ, Behar PM, Wax MK. Atypical presentations of actinomycosis. Plead Neck 1999;21(3):264-8.

(9.) Kwartler JA, Limaye A. Pathologic quiz case 1. Cervicofacial actinomycosis. Arch Otolaryngol Head Neck Surg 1989;115(4):524-7.

(10.) Brandenburg JH, Finch WW, Kirkham WR. Actinomycosis of the larynx and pharynx. Otolaryngology 1978;86(5):ORL-739-42.

(11.) Silverman PM, Farmer JC, Korobkin M, Wolfe J. CT diagnosis of actinomycosis of the neck. J Comput Assist Tomogr 1984;8(4):793-4.

(12.) Weese WC, Smith IM. A study of 57 cases of actinomycosis over a 36-year period. A diagnostic 'failure' with good prognosis after treatment. Arch Intern Med 1975;135(12):1562-8.

(13.) Sudhakar SS, Ross JJ. Short-term treatment of actinomycosis: Two cases and a review. Clin Infect Dis 2004;38(3):444-7.

(14.) Carinci F, Polito J, Pastore A. Pharyngeal actinomycosis: A case report. Gerontology 2007;24(2):121-3.

Anna M. Lipowska, MD; Michael M. Johns III, MD

From the Department of Medicine, Northwestern University Feinberg School of Medicine, Chicago (Dr. Lipowska); and the Emory Voice Center, Department of Otolaryngology-Head and Neck Surgery, Emory University School of Medicine, Atlanta (Dr. Johns). The case described in this article occurred at Emory University.

Corresponding author: Michael M. Johns III, MD, Emory Voice Center, 550 Peachtree St., NE, 9th Floor, Suite 4400, Atlanta, GA 30308. Email: michael.johns2@emory.edu
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Title Annotation:ORIGINAL ARTICLE
Author:Lipowska, Anna M.; Johns, Michael M., III
Publication:Ear, Nose and Throat Journal
Article Type:Case study
Date:Sep 1, 2014
Words:1775
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