A 61-year-old Japanese man presented with bilateral scrotal swelling noted two weeks ago. The patient had a five-year history of hypertension and DM. His current medications included amlodipine (10 mg/day), azilsartan (20 mg/day), bisoprolol (5 mg/day), linagliptin (5 mg/day), and empagliflozin (10 mg/day). His DM has been poorly controlled, and his glycated hemoglobin (HbA1c) level remained within 7-8% during the treatment period. He had no family history of TB. He was a social drinker and a non-smoker.
Two years before presenting to our service, the patient was admitted to the hospital for investigation and treatment of newly developed dyspnea and fatigability. During that hospital stay, pleuritis, pericarditis, and peritonitis were identified (Figure 1A and B). However, despite extensive investigations, including repeated AFB smear, AFB culture, and Mtb polymerase chain reaction (PCR) using sputum, blood, urine, and pleural, pericardial, and peritoneal effusions, failed to determine the etiology. However, TB was strongly suspected due to a positive interferon-gamma release assay; lymphocyte-predominant pleural, pericardial, and peritoneal effusions with high adenosine deaminase levels; pleural membrane pathology, which demonstrated epithelioid cell granuloma with Langhans giant cells and caseous necrosis. Although Mtb was not isolated, TB chemotherapy was administered due to the high likelihood of having TB and his serious illness.
The patient underwent two months of intensive phase treatment comprising INH, RIF, PZA, and EMB with supervision. This was followed by seven months of the continuation phase with INH and RIF. After the treatment regimen, marked clinical and radiographic improvements were observed (Figure 1C and D). Four months after completing chemotherapy, while the patient was asymptomatic, a low-intensity area in the prostate was incidentally found on follow-up computed tomography. Magnetic resonance imaging confirmed a prostate mass (Figure 2). Biopsy of the prostate did not confirm a malignancy, but it revealed epithelioid cell granuloma with Langhans giant cells and caseous necrosis. However, the AFB smear and culture from the prostate biopsy and urine were negative. The patient remained under careful monitoring and without changes in his status until the scrotal swelling was noted. Figure 3 summarizes the patient's clinical course timeline, including clinical events, chemotherapy, diabetes medications, and HbA1c level.
Physical examination revealed the following: body mass index, 32.9 (height 170 cm, weight 95 kg); body temperature, 37.5 C; and blood pressure, 119/79 mmHg with a regular pulse at 107 beats/min. His scrotum was bilaterally enlarged with mild tenderness and pus discharge from a scrotal fistula. Scrotal ultrasonography revealed diffusely enlarged bilateral epididymis with a heterogeneous and hypoechogenic texture (Figure 4A and B). On magnetic resonance imaging, the mass lesion showed slightly higher and lower signal intensities relative to testicular parenchyma on T1-weighted and T2-weighted images, respectively (Figure 4C and D). The results of blood and urine examination are shown in Table 1. The glycated hemoglobin was 7.5%, which suggested that the DM was poorly controlled and complicated by stage 2 nephropathy. The estimated glomerular filtration rate was decreased relative to a baseline value of approximately 50 mL/min/1.73m2. There was a mild elevation of C-reactive protein, but neither pyuria nor hematuria was observed. The urine was sterile. The AFB smear, AFB culture, and Mtb PCR of the urine and pus discharge from the scrotal fistula were negative. Bilateral epididymectomy was then performed, and pathological examination demonstrated epithelioid cell granuloma with Langhans giant cell and caseous necrosis. Although the AFB smear was negative, the tissue tested positive for Mtb PCR. AFB culture test confirmed Mtb, and the isolated strain was resistant to INF, RIF, and EB. Table 2 summarizes the results of TB investigation. Finally, a diagnosis of multi-drug resistant (MDR) TB epididymitis was made. The patient was transferred to the TB specialized hospital for further evaluation and treatment, and then chemotherapy comprising levofloxacin, ethionamide, cycloserine, para-aminosalicylic acid, and pyrazinamide was initiated.