Gingival mass in acquired immune deficiency syndrome patient: An unusual manifestation

Abstract

Non-Hodgkin's lymphoma (NHL) is designated as an acquired immune deficiency syndrome defining condition. Although uncommon, it is essential to be wary of this neoplasm since intraoral manifestations may be the first clinical manifestation of HIV disease. The gingiva is one of the rarest intraoral sites with a prevalence of 0.6%. Careful evaluation of patients presenting with solitary atypical gingival mass can lead to early detection of HIV disease. Here, we report a case of NHL manifesting as a gingival mass in a 45-year-old HIV-positive female patient.

INTRODUCTION

Non-Hodgkin's lymphoma (NHL) is the second most common human immunodeficiency virus (HIV) related tumor after Kaposi's sarcoma. The risk of getting NHL is 60 times greater in patients with HIV disease than in otherwise healthy persons.[1] NHL usually involves lymph nodes and 20–30% involves extranodal sites.[2,3] The incidence of oral manifestation of NHL is approximately 2% of all extranodal lymphomas.[3] The favored intraoral sites are the palatal mucosa and bone. The gingiva is one of the rarest intraoral sites[1] with a prevalence of 0.6%.[4]

CASE REPORT

A 45-year-old female patient reported with a complaint of swelling in maxillary left posterior teeth region since 1 month. The swelling was insidious in onset, gradually increasing in size and was preceded by extraction of upper left first and second molar due to caries and pain. The patient was under antiretroviral therapy (ART) - tenofovir, lamivudine, and nevirapine. CD4 count of the patient at baseline was 85/6% and 397/14% after 6 months of ART. Extraorally, the patient presented with nontender diffuse swelling over the left side. Left submandibular lymph node was firm, nontender, and not fixed [Figure 1]. Intraorally, a single, well-defined, nontender, soft, and compressible, gingival mass of size approximately 3 cm × 4 cm was present on the maxillary left posterior teeth region covering the first and second premolar up to occlusal level both buccally and palatally. Overlying mucosa was reddish-pink. The occlusal surface of the lesion had indentations of the corresponding mandibular posterior teeth [Figure 2]. Clinical appearance was very similar to pyogenic granuloma. As the patient was HIV positive, Kaposi's sarcoma and NHL were also considered in the clinical diagnosis. Intraoral periapical radiograph, maxillary occlusal, and orthopantomogram showed irregular bony destruction in maxillary left posterior teeth region with loss of lamina dura. No displacement or resorption of teeth was present. Medial wall, posterolateral wall, the floor of left maxillary sinus, and left lateral wall of nasal cavity were destructed. Computed Tomography of paranasal sinus and neck was suggestive of 30 mm × 31 mm × 27 mm sized, well-defined, enhancing soft tissue lesion involving left maxillary alveolar region and gingivobuccal sulcus extending into the left maxillary sinus and left nasal cavity. The lesion caused erosion of left upper alveolus and the posterolateral wall of left maxillary sinus. Few enhancing nodes were noted in left level IB, II, and largest measuring 12 mm × 7 mm [Figure 3]. Ultrasonography (USG) of the abdomen demonstrated enlarged spleen with normal echotexture. USG of the neck was suggestive of 7 mm × 5 mm size hypoechoic lymph node with loss of hila in left level II. Histopathology from biopsy tissue revealed diffusely arranged sheets of atypical lymphoid cells in a delicate connective tissue stroma containing scattered hemosiderin pigmentation [Figure 4]. Biopsy report was suggestive of NHL, high-grade malt type. Immunohistochemistry was positive for vimentin, leucocyte common antigen, AE1, CD20 (Pan B) markers suggestive of diffuse large B-cell lymphoma. Bone marrow biopsy showed marked proliferation of atypical lymphoid cells 44%. The HIV status of the patient, radiographic findings, histopathology, and immunohistochemistry confirmed the diagnosis as NHL of diffuse large B cell lymphoma type. The patient was referred to Cancer Institute for further management where chemotherapy was planned. Unfortunately, patient expired within a few days of referral.

Figure 1.

A 45-year-old female patient presenting with diffuse swelling over left side of face

Figure 2.

Well defined gingival mass over left posterior teeth region

Figure 3.

Histopathology is suggestive of atypical lymphoid cells arranged in diffuse sheets in a delicate connective tissue stroma containing scattered hemosiderin pigmentation

Figure 4.

Radiographs suggestive of irregular bony destruction in maxillary left posterior teeth region. Well defined enhancing soft tissue lesion involving left maxillary alveolus and gingivobuccal sulcus, causing erosion of posterolateral wall of left maxillary sinus and extending into the left nasal cavity can be appreciated

DISCUSSION

Although the oral involvement of NHL is rare, they are the second most common oral malignant disease after oral squamous cell carcinoma, constituting 2.2% of all malignancies of the head-neck, and 3.5% of intraoral malignancies.[2] Among the jaw lesions, the maxilla is more commonly affected than the mandible with a predilection for the posterior sites.[5,6] Oral NHL manifests itself as painless swelling, ulceration, exophytic masses, mobility or early loss of teeth, delayed healing of extraction sites, paresthesia, rapid growth, and destruction.[6,7,8,9] Intrabony involvement can mimic a toothache and can result in dental extraction and then rapid growth of tumor from the nonhealing extraction site.[9] Radiographic findings include diffuse, ill-defined areas of bone destruction, loss of lamina dura, oblique/spiking root resorption[5] concurrent with our case. Usually, the oral manifestations of NHL are secondary to a more widespread involvement throughout the body.[6] Systemic symptoms such as fever, night sweat, weight loss are noticed, and about 20% of NHL shows evidence of spleen involvement at presentation.[6] The presented case did not give a history of night sweat, fever or weight loss. Level IB, II lymph nodes and bone marrow were involved, and spleen was enlarged. Patients with disseminated diseases and those with acquired immune deficiency syndrome (AIDS) have a much worse prognosis.[8] Median survival is 34 months in immunocompetent and 9 months in HIV-positive patients.[1] In our case, owing to the HIV status of the patient and the dissemination of the disease, patient falls under poor prognosis. The intraoral NHL may show a strong resemblance to pyogenic granuloma. Clinical diagnosis may be difficult to make, especially when it exhibits primarily with solitary manifestations. The age, site of lesion, atypical clinical presentation, the absence of chronic irritation and previous history of extraction ruled out the possibility of pyogenic granuloma in this case. However in cases reporting with such atypical clinical presentation and unknown HIV status, NHL should be included in the differential diagnosis as this may be the first identifiable evidence of the disease. Early detection of the disease can result in a better prognosis. Since, the patient was a known case of HIV/AIDS and had a recent history of extraction of a tooth in the same region following which the lesion developed, differential diagnosis included malignant neoplasm namely squamous cell carcinoma, Kaposi's sarcoma, and NHL. Histopathology findings, radiographic investigations confirmed the diagnosis as diffuse large B-cell lymphoma. By knowing the signs of NHL, dentists can play an important role in the early detection of HIV disease. Patients presenting with atypical gingival mass should be screened for HIV suspecting it to be an extranodal manifestation of NHL.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

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Conflicts of interest

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