Actinomycotic pseudo-tumour of the mid-cervical region (a case report).

SS Nagral, CV Patel, PT Pathare, AA Pandit, BV Mittal 
 Department of General Surgery, K.E.M. Hospital, Parel, Bombay, Maharashtra.

Correspondence Address:
S S Nagral
Department of General Surgery, K.E.M. Hospital, Parel, Bombay, Maharashtra.

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::   Introduction

Cervicofacial actinomycosis is usually associated with infection due to pseudofungus Actinomyces israeli. This is a gram positive, non-acid fast, anaerobic organism, which typically produces small yellowish sulphur granules. Rarely other species like A. naeslundii, A. viscosus, A. odontolyticus and A. propionica are implicated. All these are harmless commensals of the oral cavity and do not exist anywhere else in nature. Secondary to mucosal trauma or dental problcms, they invade the soft tissues of the face and neck. The classic lesion is seen in the submandibular or parotid region and consists of a woody induration with or without multiple sinuses and discharging granules. The spread is usually by contiguity through the soft tissue rather than the hematogenous or lymphatic route. Sometimes a probable lymphatic spread results in isolated neck masses, which may be confused with malignant nodal masses because of similar characteristics. The picture is further confused because of no antecedent history of oral trauma or pathology as well as absence of sinuses and granules, as in the following case report.

::   Case report

RG, a 39-year-old male resident of Bombay since many years, non-smoker, non-tobacco chewer, presented with a rapidly increasing swelling in the left mid-cervical region of one month duration. The swelling was asymptomatic. There was no history of dysphagia, dysphonia, oral or dental problems. There was no history of cough, loss of weight or fever. On examination, there was a 3 x 3 cm mass in the left mid-jugular region, just anterior to the sternomastoid. It was non-tender, firm to hard and had restricted mobility. There were no scars and sinuses and the skin was free. Oral cavity and indirect laryngoscopy were normal. No other lymph nodes were palpable. Two fine needle aspiration cytologies showed just inflammatory cells. A plain X-ray chest, barium swallow, direct laryngoscopy and oesophagoscopy were all normal. The patient was given a trial of antibiotics for ten days, to which there was no response. The patient was then subjected to an excision biopsy under general anaesthesia. At operation, the mass was firm to hard and was stuck to the sternomastoid and the carotid sheath. It was dissected free and excised in toto. The cut surface revealed a fairly homogeneous appearance without any granules or necrosis. It was reported as an inflammatory lesion on frozen section. Final histopathology revealed a typical fungal colony, consisting of a pale central granule surrounded by a dense mixed inflammatory infiltrate composed of polymorphs, lymphocytes, macrophages and giant cells. The Splendore-Hoeppli reaction was seen (See [Figure:1]) The organism observed was Gram positive, acid fast negative and on staining with Gomori's silver Methenamine (GMS), showed typical filaments. The patient was given a course of tetracycline of six weeks to eradicate residual infection. A detailed dental cheek up revealed only periodontitis. The patient was totally asymptomatic without any evidence of recurrent infection at follow up.

::   Discussion

Cervicofacial actinomycosis constitutes 41-55% of Actinomycotic lesions in various studies[2]. Today it is a rare disease. In 1938 more than 200 cases were reported[3]. A series in 1975 reported detection of only 57 cases over a 40 years' period in an institution[7]. A large University record study has reported 32 cases of actinomycosis in the Cervicofacial region and has pointed out that the high incidence observed in 1935 (1 in 14800) is decreasing to 1 in 63,000 in 1982[1].

In India, actinomycosis produced by A. Israeli is very rare[5]. Few classical cases of Cervicofacial actinomycosis were reported from Jaipur. Recent reports are however rare. Desai et al[4] in 1970, reviewed 40 cases of mycetoma infection in an institution, in which there were 2 cases of A. Israeli infection of the Cervicofacial region.

Antecedent oral problems including dental caries, periodontitis, apical abscesses, gingivitis, tonsillitis, dental manipulation and maxillofacial trauma predispose to cervical infection by allowing the organisms to enter the soft tissues[1],[5]. However, this history may not be elicited in more than 40% of cases[1]. The organisms customarily spread by direct extension into the soft tissues rather than lymphatic or hcmatogenous spread. Lymph nodes are involved only if they are in the pathway of the extending, burrowing disease. Pain and fever are predominant symptoms only in 80% of cases[1]. The classic appearance of a woody swelling with sinuses of granules may be seen only in about 30 to 40% cases[1],[6]. The term “lumpy jaw” used to describe the classic lesion is derived from the appearance observed in the bovine form of the infection[6].

Isolated skin lesions in the mid or lower cervical region as in the above case are rarely documented and suggest lymphatic spread. Bennhoff[1] reported four such cases out of 32 in the Cervicofacial region. Such masses have not been reported in Indian literature. These lesions mimic a tumour mass and hence are termed as Actinomycotic pseudotumors. They may tcmporarily regress with antibiotic therapy only to reappear again[1]. The differential diagnosis of such masses includes benign or malignant neoplasm, chronic abscesses, Tuberculous nodes and chronic fungal infections. Isolated Actinomycotic granolas are also be seen in the tongue, larynx, lacrimal glands, scalp and paranasal sinuses[1].

Fine needle aspiration cytology is not useful since it shows only inflammatory cells. Rarely, it may pick up the fungal granule. Biopsy with microscopic examination and culture is the mainstay of diagnosis, especially to identify the precise organism.

The use of antibiotics has made a dramatic change in the response rates in Cervicofacial actinomycosis. According to some, penicillin still remains the drug of choice[1]. Sulphonamides, penicillin, streptomycin, tetracyclines, chloromphenicol and cephalosporin have all been shown to be effective. Indications for surgery include diagnostic biopsy especially in isolated masses, resistant fibrotic or necrotic disease and devitalised bony sequester.

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