Localised bacillary angiomatosis of the tongue

ARTICLE

A 37-year-old woman with a 6 year history of acquired immunodeficiency syndrome (AIDS) was seen for evaluation of an inflammed ulcerated lesion on the tip of her tongue which lasted for 1 month
(Fig. 1). The patient was taking prophylactic doses of gancyclovir for severe cytomegalovirus (CMV) retinitis, which had reduced her visual acuity considerably, and pentamidine inhalations for Pneumocystis carinii pneumonitis. Laboratory tests were as follows: total T cell count 1,091 mm³, CD4⁺ cells 6 mm³, CD8⁺ cells 234 mm³, CD4⁺/CD8⁺ ratio 0.02. Superficial lymph-nodes were normal. Our presumed diagnosis was of an ulcerated lesion caused by CMV or by an atypical mycobacteriosis and a punch biopsy was taken to confirm the diagnosis (Figs. 2 and 3).

Localised bacillary angiomatosis of the tongue

Microscopic examination was characterised by a lobular, vascular proliferation within the lamina propria with mild acanthosis of the overlying epithelium. The vascular lobules were composed of rounded vessels lined with protuberant endothelial cells without atypia. Adjacent to the capillaries there was a large quantity of neutrophil debris. Warthin-Starry stain showed bacteria scattered among the capillaries.

Abdominal sonography and chest X-ray were normal. When questioned about having been scratched by cats, bitten by insects or other trauma, the patient initially denied any occurrences of this kind. Then she recalled that, while they played together, her 2 year-old son often put small objects ­ which had previously been in contact with the cat ­ into his mother's mouth. A 4-week course of treatment with erythromycin 500 mg four times a day was started which led to complete recovery within 3 weeks.

Comment

Bacillary angiomatosis (BA) is a vascular proliferation caused by a gram negative bacteria (Bartonella henselae or quintana), closely related to cat-scratch disease [1-3]. The cutaneous lesions appear as single or multiple bright-red papules, nodules or as cellulitic plaques [3]. The disease may involve the gastrointestinal tract or respiratory mucosa, liver, spleen, central nervous system or lymph-nodes [4].

A prompt diagnosis of BA is important because it is a potentially fatal, opportunistic infection. We believe the case described is interesting because of the rarity of the disease and the unusual single localisation to the tongue.

In our opinion, CMV and clinical findings of atypical mycobacteriosis should be added to the list of differential diagnoses, including the more well-known pyogenic granuloma, Kaposi's sarcoma, angiolymphoid hyperplasia with eosinophilia and verruga peruana [4, 5].

REFERENCES

1. Szaniawski WK, Don PC, Bitterman SR, Schachner JR. Epithelioid angiomatosis in patients with AIDS. Report of seven cases and review of the literature. J Am Acad Dermatol 1990; 23: 41-8.

2. Dauga C, Miras I, Grimont PA. Identification of Bartonella hensenlae and B. quintana 16s rDNA sequences by branch, genus and species-specific amplification. J Med Microbiol 1996; 45: 192-9.

3. Schwartzman W. Bartonella (Rochalimaea) infections: beyond cat scratch. Annu Rev Med 1996; 47P: 355-64.

4. Walford N, Van Der Wouw PA, Das PK, Ten Velden JJA, Hulsebosch HJ. Epithelioid angiomatosis in the acquired immunodeficiency syndrome: morphology and differential diagnosis. Histopathology 1990; 16: 83-8.

5. Fagan WA, DeCamp NC, Kraus EW, Pulitzer DR. Widespread cutaneous bacillary angiomatosis and large fungating mass in an HIV-positive man. J Am Acad Dermatol 1996; 35: 285-7.

6. Le Boit PE, Berger TG, Egbert BM, Beckstead JH, Yen TSB, Stoler M. Bacillary angiomatosis. The histopathology and differential diagnosis of a pseudoneoplastic infection in patients with human immunodeficiency virus disease. Am J Surg Pathol 1989; 13: 909-20.