Kaposi sarcoma (KS) with primary effusion lymphoma in HIV infected MSM (men having sex with men) co-infected with pulmonary tuberculosis and syphilis: a case report from India

We present a case of a 30-year-old unmarried male, engaged in same-sex sexual behaviors (MSM), who wanted to enroll in an ongoing oral Tenofovir Disoproxyl Fumarate containing PrEP demonstration project. He presented at the clinic with symptoms of purple, reddish-brown patches all over the body (Fig 1 and Fig 2) and mouth (Fig 3) for the past 3–4 months. The plaques were first noticed on the trunk 4 months back and gradual spread to other parts of the body happened over the next few months. There were no symptoms suggestive of respiratory distress or gastrointestinal involvement like bleeding or obstruction. Detailed history revealed that the patient was in a same-sex relationship since the age of 15 years and had multiple sexual contacts. He reported having unprotected sexual contact with multiple male partners during the last 3 months. He did not report any blood transfusion or intravenous use of illicit drugs for recreational purposes. The participant was afebrile (37.2 °C) on examination and the heart rate, respiratory rate, blood pressure, and BMI were 78 beats/minute, 22 breaths/minute, 100/70 mmHg, and 18.5 kg/m2 respectively. No neck lymphadenopathy or jugular engorgement was observed. Bilateral pitting pedal edema was noted during the general examination. No hepatomegaly or splenomegaly was observed on abdominal examination. The result of the neurological examination was normal. Dermatological examination showed the presence of multiple, well-defined, discrete, scaly, non-tender, non-blanching, non-itchy violaceous, raised macules and patches on the face, trunk, and extremities [palmer (Fig 4), planter (Fig 5)] and oropharyngeal mucosa with size varying between 3 mm and 4 cm in diameter with the classical truncal distribution. Plaques on the trunk were showing a typical ‘Christmas tree pattern’.

Fig. 1figure 1

Purple, reddish-brown patches on the front side of the body

Fig. 2figure 2

Purple, reddish-brown patches on the back side of the body

Fig. 3figure 3

Patches inside the oropharynx

Fig. 4figure 4

Well-defined, discrete, patches on forearm and palmer surface

Fig. 5figure 5

Well-defined, discrete patches on the plantar surface

Blood and urine samples were collected for investigations. Enzyme-linked immunosorbent assay (ELISA) for HIV-1 was positive, and Rapid Plasma Reagin (RPR) test titer was 1:32. The urine sample was positive for Chlamydia trachomatis on Cartridge Based Nucleic Acid Amplification Test. The CD4 count was 226 cells per cubic millimeter of blood. Hepatitis B surface antigen and anti-hepatitis C virus antibody tests were negative. Ultrasonography of the chest revealed bilateral mild pleural effusion with a maximum thickness of 1 cm on either side. High-Resolution Computed Tomography (HRCT) revealed pleural effusion with bilateral infiltrates and was suggestive of infective etiology, most likely pulmonary tuberculosis. Cartridge-based Nucleic Acid Amplification (CB-NAAT) of sputum sample confirmed the infection as Mycobacterium tuberculosis. CSF examination showed proteins 42 mg/dl, Glucose 59 mg/dl, Total nucleated cells (predominantly lymphocytes) = 4. MRI brain showed minimal mucosal thickening in bilateral maxillary sinuses and ethmoid sinus. Baseline Haemogram and serum Biochemistry were unremarkable except for low hemoglobin levels. The serum sample was processed for Human Herpesvirus 8 (HHV-8) DNA PCR, which was found to be negative. To confirm the diagnosis of KS, a punch biopsy of the dermal lesion was taken. The section showed a dermis with multiple, irregular, jagged vascular channels extending to the subcutaneous tissue. These spaces partly surrounded pre-existing blood vessels and sweat glands at spaces. In addition, the dermis had fibro-collagenous tissue and inflammatory infiltrates composed of lymphocytes and plasma cells. A focal collection of spindle-shaped cells was also seen. The overlying epidermis showed hyperkeratosis and irregular hyperplasia. The histopathological findings confirmed the diagnosis of Kaposi Sarcoma. The patient was started on Anti-tubercular Treatment (ATT) for pulmonary tuberculosis. Highly Active Antiretroviral Therapy (HAART) was initiated after 2 weeks of initiation of ATT. After 1 month of ATT initiation, the patient developed an ATT-induced liver injury. Complete Blood Count (CBC) and Biochemistry done after 1 month of ATT initiation showed Haemoglobin = 10.6 gm/dl, Total Leucocyte Count (TLC) = 10.3 × 103/μL, Platelet count: 115 × 103/μL, Erythrocyte Sedimentation Rate (ESR) = 40 mm/hour, Mean Corpuscular Haemoglobin (MCH) = 32 pg, Mean Corpuscular Haemoglobin Concentration (MCHC) = 33.1 gm/dl, Mean Corpuscular Volume (MCV) = 97 fl, Mean Platelet Volume (MPV) = 11 fl, Total Bilirubin = 12.70 mg/dl, Direct bilirubin = 9.10 mg/dl, serum glutamic-oxaloacetic transaminase (SGOT) = 142 IU/L, serum glutamic pyruvic transaminase (SGPT) = 152 IU/L, Alkaline phosphatase (ALP) = 64 IU/l, serum creatinine = 0.67 mg/dl, serum uric acid = 16.6 mg/dl, serum urea = 15 mg/dl). Hence, he was put on liver sparing Anti Tubercular Therapy (ATT). The patient was then referred to a higher level of care for further management. He was found to have severe hypoalbuminemia leading to anasarca. During the hospital stay, he developed progressive breathlessness along with a worsening pleural effusion (left side). He was managed with repeated therapeutic thoracocentesis because of reaccumulating fluid. Cyto-morphology of the pleural fluid revealed features of primary effusion lymphoma. Flow cytometry of the pleural fluid was suggestive of PEL (large atypical cells expressing CD38, CD 56, and CD4, negative for pan B cells and T cell markers). CD 34 was negative. Video-assisted thoracoscopic surgery (VATS) could not be done due to the poor general condition of the patient. Considering the visceral involvement of the disease, systemic chemotherapy was initiated along with HAART and ATT. The prognosis of the case is poor and the same was informed to the patient in the language he understands.

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