Case report
Pharyngeal actinomycosis: a case report Francesco Carinci1, Jessica Polito2 and Antonio Pastore3 1
Department of Maxillofacial Surgery, University of Ferrara, Arcispedale S. Anna, Ferrara, Italy; 2Department of Maxillo Facial Surgery, St Anna Hospital and University, Ferrara, Italy; 3Department of Otolaryngology and Head and Neck Surgery, Ferrara University Medical Centre, Ferrara, Italy
Gerodontology 2007; 24; 121-123 Pharyngeal actinomycosis: a case report Actinomycetes are saprophytic bacteria of the oral cavity. They can produce a rare, chronic, and suppurative process that usually originates from the teeth and mandible and then involve the cervical region, especially in the sub-mandibular area. A case of actinomycosis occurring in the retropharyngeal space in a 74 year-old man is reported. It arose as a swelling behind the third lower left molar that had no lesion. The patient was treated successfully with antibiotic therapy. The clinical presentation and management of the case are discussed and the relevant literature is reviewed. Keywords: actinomycosis, actinomycetes, pharynx, neck, abscess. Accepted 1 February 2007
Introduction Actinomycosis is a chronic, suppurative and granulomatous process caused by Actinomycetes. They are usually saprophytic bacteria of the oral cavity, but sometimes can lead to local and diffuse infections. The first human case of actinomycosis was described by Von Langenbeck in 1845 and it was attributed to a fungus. Israel and Ponfick, in 1891, delineated the anaerobic nature of Actinomyces and isolated it from humans. In the 1960s Waksman showed that Actinomyces was a gram positive bacteria and in 1938, Cope first classified actinomycosis into three distinct forms: (i) cervico-facial, (ii) pulmonothoracic and (iii) abdomino-pelvic forms which are 50%, 30% and 20% of cases respectively1. Five species of Actinomycetes have been identified: Actinomycetes israeli, A. bovis, A. naeslundii, A. viscous and A. odontolyticus. These organisms are members of the family Actinomycetaceae, Stretomycetaceae, and Actinoplanaceae and except for Actinomycetes bovis, all species are normal flora of the oral cavity 2,3. Trauma in dental extraction, caries or dental manipulation are the main causes of destruction of the natural barrier and can lead to infection. Debilitating states such as diabetes, malignancy and
immunosuppression are predisposing factors. There is a slight male predominance (ranging from 1,5 to 3) and the fifth decade of life is the most affected. There is no racial predisposition or known geographic factors. The most common affected site is the cervical region, especially in the submandibular area. The diagnosis is based on cytology [fine needle aspiration cytology (FNAC)] or biopsy, and computerized tomography (CT) and/or magnetic resonance tomography (MR) help in defining the extension of the disease. The propensity of this disease to mimic carcinoma or tuberculosis is well known. The first case of actinomycosis occurring in the retropharyngeal space that was correctly diagnosed by culture examination of a specimen recovered by aspired suppurative material with FNAC is reported.
Case report A 74 year-old male was referred to the ENT Department of Ferrara University who had a history of left nucal swelling without pain for one year. The patient was a heavy smoker and drinker and was a farmer. He had had no operations in the head or neck region but referred a previous swelling behind the third lower left molar.
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Examination of the neck showed a large (4 · 6 cm), tender, fixed mass in the left cervical region, but the covering skin was normal without any sign of inflammation. A complete airway endoscopy was carried out without results. Imaging investigations were performed and sonography examination showed some enlarged lymph nodes on the left cervical side. CT identified two large cervical masses (2 cm in diameter) on the left side, one of which was localized between the left splenio and the medium scalene muscles, the other in the retropharyngeal space. There were no enlarged lymph nodes on CT images. MR confirmed the presence of the two masses on the left cervical region, both with a colliquative central area (Fig. 1). These lesions were close to the scalene and the sternocleidomastoideo muscles. Laboratory investigations showed a lymphocytosis. Serological tests (i.e. immunometry and TORCH complex) were negative for tuberculosis, bartonellosis or borelliosis infections and the chest X-rays were normal. An ultrasound-guided aspiration (FNAC) was performed on both cervical masses and the exudates contained yellow granules – known as sulphur granules – these being a hallmark of actinomycosis. On microscopic examination, colonies had a radial disposition around the abscess (Fig. 2). The therapy was benzylpenicillin 2 million units six time/die e.v. for 6 weeks. After 4 months follow-up the patient is well with no evidence of recurrence of the disease.
Figure 1 The axial MR scan showing the retropharyngeal mass filling the left inner muscles of the neck.
Figure 2 Microphotography showing the actinomyces flora stained with PAS: the colonies have a radial disposition around the abscess. Original magnification 320·.
Discussion Actinomycosis is a chronic and suppurative process that can occur in the head and neck region. Here, it usually occurs as a mass in the sub-mandibular triangle, which may be tender on palpation, associated with surrounding induration or erythema, but rarely involves the lymph nodes. A review of the literature revealed other locations for these lesions such as the larynx, middle ear and nasal septum, but no previous report has been made for the retropharyngeal space 3–6. The clinical diagnosis is difficult because its onset is not specific and the differential diagnosis cover a wide range of diseases, from tumour to chronic infections (for example carcinoma and tuberculosis). In any case, two elements will allow diagnosis to be established: positive cultures and biopsy showing the bacterium7. Sonography is routinely employed as a first imaging technique to scan the cervical region as it is inexpensive, rapid and has no side effects. It can be performed together with a sonography guided FNAC examination to collect material for a pathological report. CT is more effective at defining the extension of the abscess and the surrounding anatomical structures1,7. However, this may not be specific as it shows an ill-defined soft tissue mass in the neck with slightly less attenuation than muscle. An enhancing rim may be found when contrast is administered. The radiological differential diagnosis includes brachial cleft cyst, lymph nodes metastases, inflammatory lymph adenopathy and vascular tumor. Modern MR is the best imaging technique to define the relation between inflammatory process and cervical soft tissues and is essential for planning any surgical approach.
2007 The Authors. Journal compilation 2007 The Gerodontology Association and Blackwell Munksgaard Ltd Gerodontology 2007; 24: 121–123
Pharyngeal actinomycosis
With the advent of FNAC, the pathological diagnosis has become somewhat easier and less invasive 8. Any pus may show the typical yellow granules – colonies of organisms known as ‘sulphur granules’ – and this may help make the diagnosis. The material drained can be sent for culture, but the organism is very difficult to culture, with less than 50% being positive. On microscope examination, the colonies are seen as basophilic masses with a granular centre and a radiating fringe of club-shaped protrusions as well as the distinctive filamentous and beaded actinomyces9. Complications of cervical actinomycosis are rare, yet may be severe and life threatening10. Any spread down the bronchial tree can lead to the pulmono-thoracic manifestations including pneumonia, empyema and thoracic draining fistulas. Central nervous system involvement, either as a result of haematogenous spread or by direct extension, may lead to brain abscess, meningitis or subdural empyema. Also rupture of the carotid artery in the neck have been reported11. In the past, surgery has been used both to diagnose and to treat actinomycosis. Nowadays, diagnosis can be made with FNAC and antibiotics are the elective therapy. Surgical treatment can be performed to drain the abscess and to obtain a correct diagnosis when multiple FNACs are not diagnostic.
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2. Luckhaupt H, Arhens A. Anaerobic infections in the head and neck area. Current status of knowledge. HNO 1993; 41: 22–29. 3. Nelson EG, Tybor AG. Actinomycosis of the larynx. Ear Nose throat J 1992; 71: 356–358. 4. Belmont MJ, Behar PM, Wax MK. Atypical presentation of actinomycosis. Head Neck 1999; 21: 264–268. 5. Chang CY, Lawlwani AK, Lanser MJ. Actinomycosis of the external auditory canal. Otolaryngol Head and Neck Surg 1993; 108: 73–75. 6. Kingdom TT, Tami TA. Actinomycosis of the nasal septum in a patient infected with the human immunodeficiency virus. Otolaryngol Head and Neck Surg 1994; 111: 130–133. 7. Mabeza GF, Macfarlane J. Pulmonary actinomycosis. Eur Respir J 2003; 21: 545–551. 8. Ponifex A, Roberts F. Fine needle aspiration biopsy cytology in the diagnosis of inflammatory lesions. Acta Cytol 1985; 29: 979–982. 9. Kwartler JA, Limaye A. Cervicofacial actinomycosis: pathologic quiz case. Arch Otolaryngol head and Neck Surg 1989; 115: 524–526. 10. Podoshin L, Rosenman D, Fradis M, Wallish G. Cervicofacial actinomycosis. Ear Nose throat J 1989; 68: 559–561. 11. Cocuroccia B, Girolomoni G. Primary cutaneous actinomycosis of the forehead. JEADV 2000; 17: 331–333.
Correspondence to: Francesco Carinci, MD, Chair of Maxillofacial Surgery, University of Ferrara, Arcispedale S. Anna, Corso Giovecca, 203, 44100 Ferrara, Italy. Tel.: +39 0532.236383 Fax: +39 0532.247709 E-mail:
[email protected]
2007 The Authors. Journal compilation 2007 The Gerodontology Association and Blackwell Munksgaard Ltd Gerodontology 2007; 24: 121–123
