jabbrv-ltwa-all.ldf
jabbrv-ltwa-en.ldf
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.