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British Journal of Oral and Maxillofacial Surgery 45 (2007) 670–672

Short communication
Rare presentation of Mycobacterium avium-intracellulare
infection
Rajesh K. Alagarswamy, Wayne Halfpenny, J.K. Thiruchelvam ∗ , W. Mohamid
Barnet and Chase Farm Hospital NHS Trust, The Ridgeway, Enfield, Middlesex EN2 8JL, United Kingdom

Accepted 21 September 2006


Available online 20 November 2006

Abstract

Mycobacterium avium-intracellulare causes clinical disease mainly in immunocompromised patients with long-standing pulmonary disease,
the symptoms of which are identical to those of pulmonary tuberculosis. In patients with AIDS the infection is typically disseminated.
Extrapulmonary infection by M. avium-intracellulare is seen in children aged 1–5 years as cervicofacial lymphadenitis. Other extrapulmonary
sites are less common and include musculoskeletal, maxillary sinus, mastoid, breast, small bowel, genitourinary tract and cornea. Such
infection of the hard palate is rare. We present an unusual case of infection of the palate by M. avium-intracellulare in a 53-year-old diabetic
man. To our knowledge, this infection presenting as a palatal lump in a patient without HIV or pre-existing lung disease has not been previously
reported.
© 2006 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Keywords: Mycobacterium avium-intracellulare; Atypical mycobacteria; Palate

Case report Carcinoma, Wegener’s granulomatosis and necrotising


sialometaplasia were considered. An urgent biopsy was
A 53-year-old white man was referred with a lump in obtained along with routine haematology tests, chest, radio-
the palate of 1 month duration. It had persisted despite a graph, and fine needle aspiration cytology (FNAC) of the
course of erythromycin and metronidazole. There was no his- lymph node. One week later the patient felt unwell and
tory of dental disease, fever, cough, loss of weight, recent complained of pyrexia and general malaise. Histological
travel abroad or contact with tuberculosis. He had well- examination of the palatal biopsy specimen was reported
controlled Type II diabetes on Gliclazide, with glycosylated as showing necrotising granulomatous inflammation con-
haemoglobin C 6%. On examination, both submandibular sistent with Wegener’s granulomatosis. The FNAC showed
lymph nodes were enlarged and tender, with erythema of inflammatory cells only. Chest radiograph and blood tests
the overlying skin. There was a painful sloughing ulcer in his were within the reference ranges. Tests for antineutrophilic
mouth involving the anterior hard palate (Fig. 1). The gingival cytoplasmic antibodies and Mantoux test showed no abnor-
margins of the associated teeth were swollen and erythema- malities. In view of the systemic symptoms the patient was
tous. The teeth were vital and not mobile. Dental radiographs admitted and treated with high dose of prednisolone and Co-
showed no abnormality. amoxiclav.
Review by the physicians suggested that the history was
typical of tuberculosis with reactive enlargement of the
∗ Corresponding author at: Department of Oral & Maxillofacial Surgery,
submandibular nodes. The palate was biopsied again, and
this specimen showed non-caseating epithelioid cell-type
Barnet and Chase Farm Hospital, The Ridgeway, Enfield, Middlesex EN2
8JL, United Kingdom. Tel.: +44 208 375 2098; fax: +44 208 375 2098. granulomata (Fig. 2). These findings suggested mycobacte-
E-mail address: jkthir@yahoo.co.uk (J.K. Thiruchelvam). rial infection, despite our failure to grow acid fast bacilli

0266-4356/$ – see front matter © 2006 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
doi:10.1016/j.bjoms.2006.09.012
R.K. Alagarswamy et al. / British Journal of Oral and Maxillofacial Surgery 45 (2007) 670–672 671

culitis AFB were subsequently identified using a modi-


fied Ziehl–Neelsen stain (Fig. 3). M. avium-intracellulare
was cultured after 4–6 weeks incubation. The patient was
given clarithromycin based on the sensitivity result. His con-
stitutional symptoms resolved the palatal lesion and lym-
phadenopathy improved. In view of the risk of relapse, clar-
ithromycin was continued for 2 years.

Discussion

M. avium-intracellulare is widespread in the environment.1


However, the incidence of clinical disease in immunocom-
petent hosts is remarkably low. Patients at greatest risk of
Fig. 1. Lesion of palate with inflammation of gingival. infection are those with underlying lung disease and who have
AIDS.2 Previous gastrectomy, immunosuppressive drugs,
and diabetes mellitus are also predisposing factors. It com-
monly presents as a pulmonary infection similar to tuber-
culosis, or as cervical lymphadenitis,3 which is the usual
manifestation in otherwise healthy children under 5 years. It
may also affect unusual sites such as the wrist, mastoid, max-
illary sinus,4 but is rare in the mouth.5 It is typically indolent
and there is often a delay in diagnosis, which is usually made
by histopathological examination or microbiological stain-
ing, or culture.6 Results of cultures can take 4–8 weeks and
obtaining antibiotic sensitivities takes a further 3–4 weeks
so histological evidence of granulomatous changes should
prompt the clinician to consider mycobacterial infection.
Treatment is usually with antibiotics, or surgery or
both. It is generally resistant to most antituberculous
drugs,1,7 although extended-spectrum macrolide drugs (clar-
Fig. 2. Non-caseating epitheliod cell granulomas (haematoxylin and eosin
original magnification ×200). ithromycin and azithromycin), rifabutin, ethambutol, and
streptomycin are effective.7 Because the response can vary
on conventional stain. The steroid was therefore slowly long-term antibiotic treatment is advised to ensure complete
withdrawn. cure. Surgical excision of persistent or necrotic lymph nodes
On further review of the original slides (stained with may also be required.
haematoxylin and eosin) and sections (staines with elastin- In our case, mycobacterial infection was suspected from
Van Gieson) we found no evidence of granulomatous vas- the histopathological report and subsequently confirmed
as M. avium-intracellulare microbiologically. The patient’s
general condition and palatal lesion improved after a few
weeks’ treatment and surgical intervention was unnecessary.
Mycobacterial infection should be considered in the differen-
tial diagnosis of any patient who presents with a lesion, that
shows histological evidence of non-caseating granuloma-
tous changes with epithelioid cells, and treatment instigated
accordingly.

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