A case of mistaken identity.

Sir,

We report a case of a 29-year-old female patient who presented with a progressive growth and associated pain in the left maxillary region since 2 months and fever since 5 days. The submandibular lymph nodes were palpable, one on the right, two on the left, nontender, firm in consistency, and mobile. Intraorally, a brownish mass was present in the left maxillary gingiva occupying the buccal sulcus and surrounding the third molar [Figure 1]. The growth was sessile, lobulated, soft, and nontender. The palatal mucosa showed chronic pseudomembranous candidiasis.

Figure 1

A sessile brownish mass in the left buccal sulcus extending from maxillary first molar till maxillary tuberosity region

The total leukocyte count and platelet count were 4000 cells/mm3 and of 69,000 cells, respectively. The erythrocyte sedimentation rate was 98 mm/h, and hemoglobin was 10.4 g%. The peripheral smear showed microcytic hypochromic anemia. The TRIDOT test was positive for HIV-I. This was confirmed by HIV antibody test performed by HIV Comb and Pareekshak HIV Triline card test. No bone changes were observed in the radiographs except for soft tissue opacity in maxillary second and third molar region [Figure 2].

Figure 2

Intraoral periapical radiograph (a) and cropped panoramic radiograph (b) soft tissue opacity in the left maxillary second and third molar region with no bony involvement

An incisional biopsy with histopathology showed monomorphic, large tumor cells with eosinophilic cytoplasm and predominantly large vesicular nuclei with prominent nucleoli. Mitosis and apoptotic bodies were evident. All these features were suggestive of plasmablastic lymphoma [Figure 3].

Figure 3

Histopathological photomicrograph revealing monomorphic, large tumor cells with eosinophilic cytoplasm, large vesicular nuclei with prominent nucleoli

Immunohistochemistry was performed in which tumor cells expressed leukocyte common antigen (LCA), CD20, and B-cell lymphoma 2 (BCL2). The cells were immune-negative for CD3, CD138, and lambda light chain. The cells stained equivocal for CD10 and kappa light chain. Mib-1 proliferation was 95% approximately giving the impression of non-Hodgkin's lymphoma (NHL) or Burkitt-like lymphoma (BLL).

Topical application of clotrimazole 1% mouth paint for oral candidiasis and surgical excision of the lesion were advised. The patient was referred to ART and then to oncology centers. The patient failed to follow-up and on further contact with the parent it was learnt that the patient had declined treatment, and she survived for <2 months of her last visit to us.

BLL is an AIDS-related lymphoma which resembles Burkitt lymphoma with cells larger than the latter and with a high proliferation fraction.[1] The head and neck is the second most common region for the extranodal lymphoma, the first being gastrointestinal tract. Intraoral involvement is rare; however, the maxilla is affected more commonly than mandible, the most common site being the palate and gingiva.[2]

CD20 (B-Lymphocyte Surface Antigen B1) antigen plays a role in the regulation of human B-cell proliferation. CD20 is strongly positive in almost all B-cell lymphomas. Immunohistochemical staining of LCA is useful in identifying NHL with a high level of accuracy.[3] Proto-oncogene BCL2 encodes the BCL2 protein, which inhibits apoptosis. BCL-2 overexpressing lymphomas are basically caused due to decreased cell death rather than proliferation; hence, they tend to be slow growing than other lymphomas.[4]

MIB-1 is a monoclonal antibody equivalent to monoclonal antibody anti-Ki-67 which is used to study the proliferative index in NHL.

In the present case, the positive staining of CD20 demonstrated B cell lymphoma. BCL2 positivity and equivocal CD10 staining are suggestive of BLL. Davi et al. analyzed the characteristics of BLL within a large series of AIDS-NHL. They concluded that BLL is a frequent entity among AIDS lymphomas and should be considered as a morphologic variant of BL in the context of severe immune depression that occurs in HIV-infected patients.[5] BLL occurring in the extranodal site like gingiva is very rare. Lesions involving gingiva pose difficulty in diagnosis. The detection of the type of NHL with immunohistochemistry is essential for the treatment with relevant chemotherapy protocols to improve the survival rates of the patients.

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