Abstract
Co-infection of acute cholangitis with dengue fever and scrub typhus is
rare and life threatening. We present a case of 74-years-female known
case of chronic obstructive pulmonary disease (COPD) who had epigastric
pain, fever and vomiting for 3 days. She was found to have shock with
altered sensorium and icterus. She tested positive for scrub typhus and
dengue fever. She was managed conservatively as biliary drainage were
not done due to thrombocytopenia and patient party denial. Early
diagnosis and treatment are of paramount value in severe acute
cholangitis along with dengue fever and scrub typhus.
Keywords : Acute cholangitis, dengue, scrub typhus
Introduction
Acute cholangitis, also known as ascending cholangitis, is a
life-threatening systemic condition that results from a biliary tree
infection and obstruction.1 Severe acute cholangitis
was reported to have a mortality rate between 11 and 27% in the 1990s.1 Acute cholangitis is usually associated with
translocation of gastrointestinal flora but co infection with other
organisms is rare like scrub typhus and dengue fever. Scrub typhus is a
rickettsial disease caused by Orientia tsutsugamushi , it is
sometimes accompanied by severe systemic complications. Acute
cholangitis has been previously reported as a complication just in a
single case report but not as co infection. Although the exact mechanism
of the development of acute cholangitis and is association is unknown,
systemic vasculitis or perivasculitis caused by Orientia
tsutsugamushi has been suggested. 2 Likewise, Dengue
fever (DF) is a viral infection transmitted by vector Aedes
aegypti mosquito with primary clinical features such as high fever and
intravascular fluid and albumin leakage, which provokes pleural
effusion, hypoproteinemia, and blood hemoconcentration. However, the
incidence of abdominal pain as a clinical manifestation of DF is rare.3
Herein we report a rare and potentially fatal case of 74 years female of
acute severe cholangitis with cholelithiasis and choledocholithiasis
with dengue fever and scrub typhus co-infection.
This case report has been reported in line with the SCARE 2020
criteria.4
Case presentation
74-years-female, known case of chronic obstructive pulmonary disease
(COPD) and referred case to our centre had complains of pain abdomen and
fever for last 3 days. The pain was acute on onset, localized in
epigastric region, burning in type, radiating to back, continuous in
nature with no known aggravating and relieving factors. Pain was
associated with nausea and one episode of vomiting which was non-bilious
and non-blood stained. She also gives history of fever without
documentation of maximum recorded temperature. She was passing stool and
flatus. There was no history of loss of consciousness, any trauma, and
weight loss. On her past medical history, she was diagnosed with COPD.
She hasn’t gone any surgical intervention in the past. Her family
history and allergic history were non-significant. She is non- smoker,
doesn’t consume alcohol and has normal bowel habits. On clinical
examination and assessment, the patient looks critically ill with
icterus, dehydration, irrelevant talk with tenderness and guarding at
epigastric region.
On examination her vital parameter showed low blood pressure (70/40
mmHg) and increase in pulse rate (146 beats/min) with low blood glucose
level of 40 mg/dl. Laboratory analysis showed leucocytosis of 66,500
cells/cumm, thrombocytopenia with 26,000 cells/cumm, elevated urea and
creatinine: 91/2.1 mg/dl and total bilirubin/direct bilirubin : 4.7/2.7
mg/dl. Arterial blood gas analysis interprets metabolic acidosis.
Patient was tested positive for dengue infection and IgM Positive for
Scrub typhus. Serology was non-reactive for HIV, Hepatitis B and C .
Ultrasonography of abdomen and pelvis was ordered which showed mild
intrahepatic biliary radicals dilatation with multiple cholelithiasis;
largest measuring 5.4 mm, and common bile duct is dilated- 14.2mm with
multiple calculi largest measuring 6.4mm.
Immediately 50 ml 25% dextrose was given as patient was in
hypoglycemia. Her blood pressure was also in lower side with tachycardia
thus fluid resuscitation with 2 pint Normal saline fast was given.
Antibiotics tazobactam and piperacillin was also administrated in
emergency ward of our centre.
With prior information to anaesthesia team for further management and
future need of centre line insertion, patient was immediately shifted to
surgical intensive care unit (SICU). Patient was kept in close
monitoring with nil per oral (NPO) with intravenous fluids. Do not
resuscitate (DNR) consent was given by the patient party at the day of
admission. Injection meropenem along with doxycycline was started at the
same day. Inotropic support with injection nor adrenaline was started to
maintain mean arterial pressure >65mmHg and urine output
> 0.5 ml/kg/hour. Four-pint platelet rich plasma (PRP) was
transfused for low platelets count after which counts elevated from
25000 to 40000 cells/cumm. With a thought of need of multidisciplinary
team approach and care for regaining optimal health of patient gastro
medicine and physiotherapy consultation was done. Central venous line
was secured by modified seldingers technique on first day of admission
after transfusion of platelets.
Daily chest physiotherapy and incentive spirometry was advised.
Injection antibiotic vancomycin and hydrocortisone was added. Daily
renal function test (RFT) was done and gradually acute kidney injury
resolved. There was a issue of hypokalemia in between which was
corrected by potassium supplementation. During stay in SICU
neuropsychiatry consultation was done for irrelevant talk and
disorientation which was managed with tab quetiapine.
Patient was allowed to have liquid diet from evening of second day of
admission. Daily complete blood count (CBC) shows decrease in total
count from 65000 cells/cumm to 26000 cells/cumm at day third of
admission in SICU. With the gradual clinical improvement in patient
health status she was step down to high care unit from surgical ICU
where she was continued with iv antibiotics but after adjustment and
continued chest and limb physiotherapy with daily vitals monitoring.
Daily laboratory reports showed gradual improvement in total counts and
other blood parameters as well as clinically better thus was planned to
discharge on oral antibiotics.
Patient was asymptomatic and hemodynamically stable with normal
laboratory parameters at the time of discharge. Patient was advised for
follow up after 2 weeks for ERCP, however patient was lost to follow up.
Discussion
Acute cholangitis occurs when biliary stenosis results in cholestasis
and biliary infection.5 Biliary obstruction leads to
break down of defensive mechanisms of biliary tree. Intraductal pressure
increases due to bile stasis which causes widening of tight junctions,
malfunction of Kupffer cells, decrease in local production of IgA,
cholangiovenous reflux allowing pathogens to access intrahepatic
canaliculi in addition to hepatic veins and lymphatics. This leads to
bacteraemia, endotoxemia and ultimately a systemic inflammatory
response.1
Potential causes of biliary obstruction include choledocholithiasis,
pancreatic cancer, metastatic tumour, primary sclerosing cholangitis,
mirizzi syndrome, roundworm or tapeworm infestation of the bile duct,
stricture of bilioenteric anastomosis, biliary stent obstruction,
amyloidosis, AIDS cholangiopathy and post-ERCP-associated acute
cholangitis. The most common cause of acute cholangitis is
choledocholithiasis accounting 50% of the cases and second common cause
being malignant obstruction accounting for 10% to 30% of
cases.6
According to the TG18/TG13 diagnostic criteria, a diagnosis of AC can be
made if the patient presents with the three pathologies of systemic
inflammation (must be present), cholestasis, and bile duct lesions (from
imaging findings).5
This was a rare case of severe acute cholangitis with co-infection of
scrub typhus and dengue fever. Dengue infections may present as true or
apparent surgical acute abdomen, sometimes misleading the
surgeons.7,8 True acute abdomen may develop as a
complication of dengue fever like ruptured splenic hematomas, upper and
lower gastrointestinal bleeding, and abdominal wall hematomas due to
coagulopathy. Furthermore, dengue infections may occur with
simulataneous surgical disease as dual pathology either
pathophysiologically unrelated to dengue (like perforation of a hollow
viscus) or pathophysiologically related to it (like acute acalculus
cholecystitis related to dengue).8 Likewise, our case
presented as acute abdomen who was found to have choledocholithiasis
induced acute cholangitis which is pathophysiologically unrelated to
dengue infection. Our patient had thrombocytopenia with platelet count
of 26000 /cumm, renal dysfunction with creatinine 2.1mg/dl, hypotension
requiring nor-epinephrine, and disturbance of consciousness categorizing
the case into severe acute cholangitis according to TG18/TG13 severity
assessment criteria.5 Charcot’s triad and Reynold’s
pentad of acute cholangitis were both present in our case which are
specific tool but not sensitive.6 Severity of acute
cholangitis in our case was contributed by both dengue and scrub typhus
infection. Only two cases of dengue and scrub typhus coinfection has
been reported from Nepal till date.9,10
Abdominal ultrasonography is first-line diagnostic study because of its
affordability, availability, and high specificity for detection of bile
duct dilation and bile duct stones but its sensitivity is insufficient.
Ultrasound findings suggestive of bile duct stones include direct
visualization of a stone or filling defect, biliary tract dilation, or a
dilated common bile duct greater than 8 mm.5,6 Our
case had all the above findings.
Urgent biliary tract drainage in patients with severe acute cholangitis;
however, it was not done in our patient because she was hemodynamically
unstable and was on DNR status. So, she was planned for ERCP once the
acute illness resolved.11
Acute cholangitis can present with co-infection of dengue fever and
scrub typhus leading to rapid deterioration of the hemodynamic condition
especially in patients from endemic regions. Early diagnosis and
treatment are of paramount value in severe acute cholangitis along with
dengue fever and scrub typhus.