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Case Presentation

A 35-year-old man presented with a month of fluctuating fever accompanied with progressive icterus. He was referred to our hospital due to the primary complaints of myalgia, arthralgia, epigastric pain, itching, and weakness, which had worsened over the last three days. He also experienced anorexia, a slight epigastric pain but had no nausea, vomiting, or productive cough.  He also complained of a recent red colored urine, dysuria, and urine frequency. No history of acholic stool was declared, or similar previous complaints. He had a past medical history of autosomal dominant polycystic kidney disease (ADPKD), which was also present in his father, sister, and brother. He had no recent travel history, no employment in paddy fields, no engagement in high-risk sexual behavior, no use of any kind of medication, no drug abuse, no raw dairy ingestion, and no alcohol consumption or smoking. He had recently started taking opium as a painkiller.  On examination, he was febrile with temperature of 39°C, tachycardia (109 beats/min), and normal respiratory rates (17/minute). His blood pressure was 125/75 mmHg. Physical examination showed was quite evident icteric sclera in addition to skin jaundice on his abdomen, a midline scar was detectable due to a laparotomy procedure performed seven years ago for an appendectomy. He had slight epigastric tenderness and Hackett’s grade 3 splenomegaly with the spleen expanding to the umbilicus. A mild unspecific hepatomegaly was also present. However, no other signs of chronic liver failure or portal hypertension such as ascites, spider angioma, or gynecomastia. Other systemic examinations such as skin, and neurologic were unremarkable. Initial laboratory tests showed no abnormalities in blood cell counts with retic count of 0.7. Biochemical tests showed a total serum bilirubin of 10 mg/dL, a direct serum bilirubin of 8 mg/dL, AST97 U/L, ALT157 U/L, ALP 495 U/L, and GGT 27 U/L. He had Serum creatinine (Cr) of 1.53 and urea of 47, in addition to many RBCs in urine analysis were reported. Amylase and lipase were both in normal range. The other hematologic parameters were as follows: PT 13, PTT 26, ESR, 59 mm/h, CRP 2+. Viral hepatitis markers were all negative (Anti-hepatitis C virus antibody (Anti-HCV), Anti-hepatitis A virus antibody (Anti-HAV (total)), Anti-HAV (IgM/ IgG), hepatitis B surface antigen (HBsAg), and anti-Hepatitis B core antibody (anti-HBc) (total/IgM/IgG)). The patient’s initial laboratory findings are reported in Table 1. In upper abdominal ultrasonography, minimal increase in liver size (160mm), and huge splenomegaly with accessory spleen (132 mm), dilated CBD (7.5mm), dilated portal vein (17mm), ADPKD, renal stones, and hemorrhagic cysts measuring 55mm in right and 36mm in left kidneys were reported. Abdominopelvic computed tomography (CT) scan without contrast confirmed the findings of ultrasonography in addition to absence of dilatation in intra and extra hepatic ducts, ruloing out malignancies and hepatobiliary stenosis. High Resonance CT (HRCT) revealed unspecific longitudinal consolidation collapse at the base of the lungs. According to clinical, laboratory and sonographic findings of dilated CBD, magnetic resonance cholangiopancreatography (MRCP) was performed and showed normal findings. We evaluated further causes of infectious hepatitis. Regarding high geographical prevalence of leptospirosis IgG and IgM, in addition to HIV antibody, tuberculin test, and VDRL were all checked and negative. Then, we conducted the wright, Coombs-wright, and 2ME test, which initially showed a titer of 1/640, 1/640, and 1/160, respectively on the second day of admission. Simultaneously, blood culture grew Brucella melitensis . According to the patient’s positive Coombs-Wright test, high levels of liver enzymes, and the history of sudden exacerbating systemic symptoms, we made a diagnosis of Brucella hepatitis. Subsequently, he was started on streptomycin 1 mg/ IM daily for the first 14 days, followed by the preferred first-line drugs, a doxycycline-aminoglycoside combination for six weeks. After a week of follow-up, general condition was improved, fever subsided, and symptoms were relieved. After a month, significant improvement in laboratory findings were also present, all signs and symptoms were gone, however, conjugated hyperbilirubinemia remained untreated (Table 2). Thus, secondary work-ups were performed to examine concomitant autoimmune hepatitis, Wilson’s disease, hemochromatosis, alhpah1-anti trypsin deficiency, rheumatologic diseases, and multiple myeloma. Ceruloplasmin, α-1 Anti Trypsin, Anti LKM Ab, Anti EMA (IgG, IgM), ASMA, AMA, ANA, P-ANCA, HLA-B51and blood protein electrophoresis were all normal. Additionally, we asked about possible hepatotoxic agents. The patient’s history of no drug intake, herbals or OTCs, but routine and very low dose consumption of painkillers was completely reliable, ruling out the drug-induced liver injury. Finally, with the possible diagnosis of DJS, he underwent a liver biopsy. The lobular liver structure was normal during Hematoxylin and Eosin (H&E) staining, however, vivid dark brown pigments in the hepatocyte cytoplasm was found, confirming the diagnosis of DJS in concomitant with brucella hepatitis (Figure 1). The Periodic Acid-Schiff-diastase (PAS-D) method was negative for alpha-1-antitrypsin globules. The patient was insured and underwent follow-up, and after one year, his condition returned to normal.