Actinomycosis of the tongue a diagnostic dilemma

Actinomycosis of the tongue a diagnostic dilemma

British Journal of Oral and Maxillofacial Surgery (1989) 27, 249-253 0266 4356X/89/0027-0249/$10.00 0 1989 The British Association of Oral and Maxill...

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British Journal of Oral and Maxillofacial Surgery (1989) 27, 249-253 0266 4356X/89/0027-0249/$10.00 0

1989 The British Association of Oral and Maxillofacial Surgeons

ACTINOMYCOSIS

OF THE TONGUE

A DIAGNOSTIC

I. D. BRIGNALL, B.D.S. and M. GILHOOLY, Department

DILEMMA B.D.s., F.D.s.R.c.s., M.R.c.s.,

of Oral and Maxilla-Facial Surgery, East Grinstead

L.R.C.P.

Queen Victoria Hospital,

Summary. Actinomycosis of the tongue is an uncommon form of this well known but little seen bacterial disease. A review of the literature is presented and a report of a case which is believed to be the first from the British Isles in recent years. The clinical appearance mimicked a deep seated malignant tumour with induration, limitation of tongue movement and pain. A Tru-Cut biopsy was obtained which revealed a diagnosis of actinomycosis. Treatment was with oral phenoxymethylpenicillin for 3 months.

Introduction

Actinomycosis is first thought to have been recognised in humans in about 1845 by Von Langenbeck (Bronner & Bronner, 1971). In 1885 Von Hacker described a case of actinomycosis of the tongue (Cameron, 1932). In humans, the causative organism is generally Actinomyces israeli though other actinomycetes (A. naeslundii, odontolyticus, viscosus and possibly bovis) may contribute. Of the body sites involved; cervico-facial, pulmonary and abdominal, the former is said to be the most common (25-65%) though only about 3% involves the tongue (Cope, 1938). In our review of the English speaking literature we were able to find only six reported cases of tongue involvement in the past 20 years (Sodagar & Kohout 1972; Uhler & Dolan, 1972; Leafstedt & Cieeson, 1475; Kuepper & Harrigan 1979; Laforgia & Mangini, 1979) although not one from the British Isles. Case report

A 55-year-old Caucasian male presented with a swelling of the left side of his tongue (Fig. 1). He reported that about 2 weeks previously he had lost normal movement of the tongue which then improved spontaneously until 3 days prior to presentation when he had experienced a sudden onset of swelling and acute discomfort of the tongue with associated dysphagia. This discomfort was sufficient to prevent sleep. A history of an accidental self-inflicted bite to the tongue was recorded 6 months previously. Examination revealed a pale, tired looking man with an upper respiratory tract infection. Current medication was allopurinol and azapropazone for gout and Magnapen prescribed by his medical practitioner for the tongue lesion 3 days previously, Extra-oral examination was unremarkable and did not reveal any lymphadenopathy. Intra-orally a tense non-ulcerated indurated swelling 2 to 3 cm in diameter was noted situated in the postero-lateral aspect of the anterior two-thirds of the tongue. Whilst the swelling was most obvious on the dorsal surface, the induration (Received 16 December

Correspondence

1987; accepted 9 August 1988)

to: Mr I. D. Brignall, 185 High Street, Tonbridge, Kent.

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Fig.

\ Figure

l-Swelling

SURGERY

1

of left side of tongue

on presentation.

extended both to the ventral surface of the tongue and the floor of the mouth. The mucosa appeared of normal colouration and whilst there was no paraesthesia, tongue movement was restricted. The clinical impression formed at the time was of a malignant tumour. Haematological examination revealed a neutrophilia and a raised ESR of 33 Westergren units. A chest radiograph was reported as normal. A series of three Tru-Cut biopsies were taken of the lesion under local analgesia in order to reach the deeper tissues with minimal trauma and obtain a representative section (Fig. 2). Histopathological examination using haematoxylin and eosin, PAS and Gram stains was carried out on the biopsy specimen and a diagnosis of actinomycosis was reported. No culture was obtained. Treatment was commenced with 500 mgs phenoxymethylpenicillin four times

Fig. Figure

2-The

Tru-Cut

needle

showing

2 biopsy

specimen

from within the lesion.

ACTINOMYCOSIS

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daily. Two months following the commencement of therapy the lesion had apparently resolved and the tongue was clinically normal. Antibiotics were continued for one further month. To date, over a year later, no recurrence has been noted. Discussion Actinomycosis of the tongue generally presents as a tender or non-tender indurated swelling usually of a few weeks duration thpugh it may also present as a small nodule or even ulcer (Cope, 1938). If untreated, this may progress to abscess formation with discharging sinuses usually on the dorsum of the tongue but rarely tracking down to the submental region; yellow ‘sulphur granules’ (colonies) may indeed be seen on examination of the pus. No lymphadenopathy is associated. Often, there may be limitation of tongue mobility due to muscular involvement (Fig. 3). Frequently, there is a history of trauma to the tongue some months previously. The case reported serves to illustrate the importance of biopsy as part of the diagnostic process of any unknown lesion of the oral cavity. Cope (1938) interestingly cites one case in which a biopsy revealed actinomycosis with pus in a sarcomatous swelling of the tongue. The Tru-Cut method used for the biopsy provides a relatively atraumatic way of reaching deeper tissues in a lesion such as the one presented, although a number of specimens are probably necessary to obtain a representative picture. The alternative method of simple incisional biopsy of such a lesion would invariably be superficially destructive given the depth of biopsy necessary. If a haemangioma is seriously considered as a possible diagnosis, then fine needle aspiration should be undertaken prior to any biopsy so as to avoid the possibility of a severe haemorrhage. Ideally, histopathological examination should be backed up by culture of the organism to give absolute confirmation though this was unfortunately not possible in our. case.

Fig. Figure

SPhotomicrograph

3

of muscle fibres with collections around a colony of actinomyces.

of acute and chro& (H&Ex320).

inflammatory

cells

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Table I Differential

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Inflammatory Actinomycosis Nocardiosis Botryomycosis Lingual Abscess

(Peabody & Seabury, (Small & Kobernick, (Luker, 1985)

1960) 1967)

Neoplastic Malignant

Primary-Sarcoma Secondary-Carcinoma

Benign

Connective Tissue Tumours including: Granular Cell Myoblastoma, Neuroma, Neurilemoma, Lymphangioma, Haemangioma

Although treatment nowadays is generally with oral phenoxymethylpenicillin (Hensher & Bowerman, 1985) which was indeed successful in this case, a number of antibiotics have been shown to be effective. Amoxycillin (Martin, 1984); minocycline (Martin, 1985); tetracycline (Bramley & Orton, 1960); or clindamycin (Leafstedt & Gleeson, 1975) provide the best alternatives. However, contrary to Hensher and Bowerman’s views that no resistance to Penicillin V has been noted, Bramley and Orton found four out of 11 cultures to be resistant to this drug in vitro but all sensitive to tetracycline. It is worth noting (Martin, 1984) that amoxycillin has generally more favourable minimum inhibitory concentration (MIC) values against Actinomyces isruefii (0.8 yg/ml) than benzyl penicillin (0.06-6.0 yglml) and this, coupled with it’s better oral absorption than Penicillin V, suggests that amoxycillin may be a better first choice therapy. The dose suggested is 500 mgs three times daily. Parenteral therapy is probably not necessary for tongue lesions unless they are exceptionally severe. Cessation of therapy on apparent resolution of the swelling may result in almost immediate recurrence, therefore treatment should be continued for a number of weeks post resolution. One reason for the slow response to treatment may be the relative avascularity of the affected tissues allowing little antibiotic penetrance which is further worsened if abscess formation supervenes (Harvey et al., 1957). The same authors consider the surgical excision of the chronic lesion to be necessary prior to the use of parenteral antibiotic therapy. A possible differential diagnosis of such a lesion is shown in Table I. Acknowledgements We would like to thank Mr Michael Awty for allowing us to present this report on his patient and Mr Peter Banks for his helpful advice in its preparation. Our thanks are also due to Dr William Brander for his pathology report and Mr Andrew Hack for photography.

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References Bramley, P. & Orton, H. S. (1960). Cervico-facial actinomycosis. A report on eleven cases. Brirish Dental Journal,

109, 235.

Bronner, M. & Bronner, M. (1971). Actinomycosis. 2nd Ed. Bristol: J. Wright & Son Ltd. Cameron, 0. J. (1932). Primary actinomycosis of the tongue with a report of two cases, Journal of the American

Medical Association,

99, 1146.

Cope, Z. (1938). Actinomycosis. Oxford University Press. Harvey, J. C., Cantrell, .I. R. & Fisher, A. M. (1957). Actinomycosis, Annals of International Medicine,

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Hensher, R. & Bowerman, J. (1985). Actinomycosis of the parotid gland. British Journal of Oral & Maxilla-Facial

Surgery,

23, 128.

Kuepper, R. C. & Harrington,

W. F. (1979). Actinomycosis of the tongue. A case report. Journal of

Oral Surgery, 31, 123.

Laforgia, P. & Mangini, F. (1979). Nodular isolated actinomycosis of the tongue. A case report. Dental Surgery,

55, 48.

Leafstedt, S. W. & Gleeson, R. M. (1975). Cervico-facial actinomycosis. American Journal of

Surgery,

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Luker, J. (1985). A case of lingual abscess. British Dental Journal, 159, 300. Martin, M. V. (1984). The. use of oral amoxycillin for the treatment of actinomycosis. British Dental Journal,

156, 252.

Martin, M. V. (1985). Antibiotic treatment of cervico-facial actinomycosis for patients allergic to penicillin: a clinical and in vitro study. British Journal of Oral & Maxillo-Facial Surgery, 23, 428. Peabody, J. W. Jnr. & Seabury, J. H. (1960). Actinomycosis and nocardiosis-A review of the basic differences in therapy. American Journal of Medicine, 28, 99. Small, I. A. & Kobernick, S. (1967). Botryomycosis of the tongue. Oral Surgery, Oral Medicine, Oral Pathology 24, 503.

Sodagar, H. & Kohout, E. (1972). Actinomycosis of the tongue as a pseudotumour.

Laryngoscope,

2149.

Uhler, I. V. & Dolan, L. A. (1972). Actinomycosis

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82,