Case Report: Biloma gastrostomy after failed sonogram-guided percutaneous aspiration, pigtail catheter insertion and surgical drainage

Abstract

Bilomas are rare abnormal extrabiliary accumulation of bile. This can be either intrahepatic or extrahepatic following traumatic or spontaneous rupture of the biliary tree. The commonest causes of biloma are surgery, percutaneous transhepatic cholangiography, percutaneous transhepatic biliary drainage, transcatheter arterial embolization and abdominal trauma. We report here a 15 year old patient whom we followed for over 10 years. His chief complaints were right hypochondriac pain, loss of appetite and vomiting. Initial clinical presentation, sonographic as well as laboratory findings suggested a liver abscess, which was drained, but the definitive diagnosis of biloma was entertained after sonographically guided percutaneous aspirations and percutaneous transhepatic cholangiography 7 years later. We also discuss the role of imaging and surgical challenges encountered that culminated into bilomo-gastrostomy. The patient is now enjoying a peaceful life.

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CASEREPORT
Case Report: Biloma gastrostomy after failed sonogram-guided
percutaneous aspiration, pigtail catheter insertion and surgical
drainage [version 1; referees: awaiting peer review]
TomR.Okello ,   DavidsonOcen , JimmyOkello , IrenePecorella ,
DerrickAmone 6
DepartmentofSurgery,StMary’sHospitalLacor,Gulu,Uganda
GuluUniversity,Gulu,Uganda
DepartmentofAnesthesia,StMary’sHospitalLacor,Gulu,Uganda
DepartmentofRadiology,StMary’sHospitalLacor,Gulu,Uganda
DepartmentofRadiological,OncologicalandAnatomicPathologySciences,SapienzaUniversityofRome,Rome,Italy
DepartmentofSurgery,GuluUniversity,Gulu,Uganda
Abstract
Bilomasarerareabnormalextrabiliaryaccumulationofbile.Thiscanbeeither
intrahepaticorextrahepaticfollowingtraumaticorspontaneousruptureofthe
biliarytree.Thecommonestcausesofbilomaaresurgery,percutaneous
transhepaticcholangiography,percutaneoustranshepaticbiliarydrainage,
transcatheterarterialembolizationandabdominaltrauma.Wereportherea15
yearoldpatientwhomwefollowedforover10years.Hischiefcomplaintswere
righthypochondriacpain,lossofappetiteandvomiting.Initialclinical
presentation,sonographicaswellaslaboratoryfindingssuggestedaliver
abscess,whichwasdrained,butthedefinitivediagnosisofbilomawas
entertainedaftersonographicallyguidedpercutaneousaspirationsand
percutaneoustranshepaticcholangiography7yearslater.Wealsodiscussthe
roleofimagingandsurgicalchallengesencounteredthatculminatedinto
bilomo-gastrostomy.Thepatientisnowenjoyingapeacefullife.
Keywords
Biloma,imaging,bilomagastrostomy,Ultrasound.
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Referee Status: AWAITING PEER
REVIEW
05Jul2018, :19(doi: )First published: 1 10.12688/aasopenres.12876.1
05Jul2018, :19(doi: )Latest published: 1 10.12688/aasopenres.12876.1
v1
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AAS Open Research 2018, 1:19 Last updated: 05 JUL 2018

AAS Open Research

TomR.Okello( )Corresponding author: okellotomrich@gmail.com
 :Conceptualization,DataCuration,FormalAnalysis,Investigation,Methodology,ProjectAdministration,Resources,Author roles: Okello TR
Software,Supervision,Validation,Visualization,Writing–OriginalDraftPreparation,Writing–Review&Editing; :DataCuration,Ocen D
Investigation,Methodology,Resources,Writing–OriginalDraftPreparation,Writing–Review&Editing; :Investigation,Methodology,Okello J
Resources,Writing–OriginalDraftPreparation; :Investigation,Methodology,Resources,Software,Supervision; :DataPecorella I Amone D
Curation,FormalAnalysis,ProjectAdministration,Resources,Software,Validation,Visualization,Writing–OriginalDraftPreparation,Writing–
Review&Editing
Nocompetinginterestsweredisclosed.Competing interests:
OkelloTR,OcenD,OkelloJ How to cite this article: et al. Case Report: Biloma gastrostomy after failed sonogram-guided percutaneous
AASOpenResearch2018, :19aspiration, pigtail catheter insertion and surgical drainage [version 1; referees: awaiting peer review] 1
(doi: )10.12688/aasopenres.12876.1
©2018OkelloTR .Thisisanopenaccessarticledistributedunderthetermsofthe ,whichCopyright: et al CreativeCommonsAttributionLicence
permitsunrestricteduse,distribution,andreproductioninanymedium,providedtheoriginalworkisproperlycited.
ThisworkwassupportedbytheAfricanAcademyofSciencesthroughaDELTASAfricaInitiativeGrant[DEL-15-011],aspartGrant information:
oftheTHRiVE-2initiative.
The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.
05Jul2018, :19(doi: )First published: 1 10.12688/aasopenres.12876.1
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Introduction
Biloma is a rare but challenging condition in Uganda and
Sub-Saharan Africa. The term biloma was introduced in 1979 by
Gould and Pater to describe a loculated bile collection located
outside the biliary tree1. Lee and Suh (2007), also defined
biloma as loculated collection of bile outside the biliary tree2.
As an encapsulated collection of bile within the abdomen, a
biloma is formed when there is bile duct interuption3. Bilomas
are often found during the postoperative period many days after
surgery as a localized encapsulated extraductal bile collection4.
Post-operative biliary leaks are rare complications of abdomi-
nal surgery, but if untreated, may result in significant morbidity
and mortality5. Although bilomas are usually a result of surgical
complications and abdominal trauma, spontaneous bilomas also
do occur, but these are rare6.
Case report
The patient first presented to St Mary’s Hospital Lacor, Gulu
district in July 2007 at 15 years of age with complaints of right
hypochondrial pain associated with vomiting and loss of appetite
for 2 months. There was no abdominal distention, no constipation
and no fever. The patient was found to be afebrile, not jaundiced,
not anemic and had normal blood pressure. His abdomen was of
normal fullness and soft, but he had a tender, enlarged smooth
edged liver 10 cm below the costal margin. The patient’s eryth-
rocyte sedimentation rate was elevated at 70 mm/h, and he
exhibited bleeding and clotting times of 4 min and 4 min 30 s,
respectively (normal range is 1–8 mm/h for adults); other blood
indices were within normal range. A stool examination showed
no ova or cysts, HIV serology was negative and liver enzymes
were slightly elevated.
Ultrasound (US) of the abdomen revealed an echo-complex mass
in the liver hilum extending to the left lobe of the liver. A diagno-
sis of liver abscess to rule amoebic liver abscess was entertained
(Figure 1a, b). Approximately 1100 ml pus was percutaneously
aspirated under US guidance and thereafter serial percutaneous
aspiration was performed every 48 hours and antibiotics were
prescribed for 10 days, IV ciprofloxacin (500 mg every
12 hours) and IV metronidazole (500 mg every 8 hours). This
treatment did not elicit a response, thus a surgical incision
was made and drainage was performed on the 11th day and a
drainage tube was left in situ. The patient was discharged after
significant improvement
After 25 months (18 years of age), the patient presented with
similar complaints of pain in the right hypochondrium and a
tender right upper quadrant. The findings of respiratory, cardio-
vascular and neurological examinations were unremarkable. US
results showed a recurrent liver abscess measuring 8×10 cm and
the patient had normal bleeding and clotting time. A percutaneous
pigtail catheter was placed in situ and after emptying 277 ml pus,
the patient was discharged for review after 2 months.
The patient continued to present with complaints of epigastric
pain, progressive weight loss, easy fatigability, due to recurrent
liver abscesses, and percutenous drainage was the preferred mode
of therapy. After a further 3 years (at 21 years of age) he developed
fever and, worsening epigastric pain, with no vomiting and
no constipation. During this particular episode the patient’s
appearance indicated illness. The patient had a body tempera-
ture of 39°C, a blood pressure of 131/74 mmHg and a pulse of
80 bpm. His abdomen had right hypochodrial tenderness. US and
percutenous aspiration revealed 420 ml yellowish bilious fluids
mixed with pus. Culture and sensitivity analysis of the aspirate
reveal Streptococcus pyogenes sensitive to ciprofloxacin and
chloramphenicol. Percutenous serial aspiration with a 2-week
course of the aforementioned antibiotics using the same regimen
led to a significant improvement. In September 2016, a percute-
nous transhepatic cholangiogram (PTC) revealed a bile lacunae
and a diagnosis of biloma was made, but on the fifth day after the
PTC, the patient developed cholangitis due to Staphylococcus
aureus, which responded to antibiotics. A follow-up sonogram
revealing the biloma is shown in Figure 2a, b.
Figure 1. Abnormal ultrasound findings. (a) Abdominal Sonogram
in transverse (left) and sagittal (right) sections showing an echo-
complex fluid collection sub-hepatic left lobe liver, 10×8 cm in size.
(b) Sonogram obtained 3 months following ultrasound-guided serial
aspiration in transverse (left) and sagittal (right) sections show an
echo complex indicating fluid collection in the sub-hepatic left lobe
of the liver. The size of fluid collection had reduced to 277.1 cm3 in
volume.
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performed as an internal drainage procedure. The post-operative
recovery was uneventful and the patient was discharged on
day 9 after surgery. Since then, up until the point of writing, the
patient has been symptom-free and continues to enjoy good life
and gainful employment.
Discussion
Bilomas are rare, with a reported incidence of about 2.5% after
cholecystectomy6. The prevalence of biloma after hepatectomy
is 35.7% (95 % CI, 26.2–45.2), but after blunt liver injury it
rises to 36%7. Post-traumatic and post-surgical collections of
encysted bile (bilomas) can be difficult to diagnose, such that
puncture of the cystic lesion under radiological guidance is
essential8. Spontaneous biloma caused by spontaneous rupture
of the intrahepatic duct, without any underlying disease (as
occurred in our patient) is a very rare finding2. Bilomas can
become infected2.
Clinically, patients with bilomas present with right upper
quadrant pain, fever within 7 days and in rare situations with gas-
tric outlet obstruction6. Patients with biloma will present with
abdominal pain, nausea, anorexia, jaundice, and fever. How-
ever, this may vary from minimal symptoms to full blown
biliary peritonitis9. Presentation of right upper quadrant
abdominal pain associated with a history of recent abdomi-
nal trauma or surgery is suspicious and diagnosis is confirmed
by detection of typical radiological features, with a differen-
tial diagnosis of a pseudocyst10. CT scans and MRI revealing
upper abdominal fluid collection may aid the diagnosis of
biloma3.
Biloma management may vary, from percutaneous aspira-
tion, percutaneous catheter drainage, surgical drainage to overt
surgical treatment; however, smaller bile leaks often resolve
spontaneously in few days9. A drainage catheter positioned
and left in place often leads to improvement after 7 days11.
Binmoeller et al. report that endoscopic treatment of biloma is
technically successful in 95% of cases5. However, biloma occur-
ring after hepatectomy and after blunt liver injury is self-limiting12.
Endoscopic decompression main biliary tract could be often useful
in treating biloma13. Biloma are also often successfully treated with
Endosonography (EUS)-guided biloma drainage14.
Whilst, Chen, Geng and Zhao (2002)15, and other studies13,16
report that there is no need for surgical exploration, since biloma
is self limiting7 and in other situations percutaneous insertion
of a drainage catheter or simple needle aspiration are adequate2,
other authors allude to surgical drainage for biloma6. Transgastric
drainage of biloma has been found to successfully treat patients
with biloma achieving complete fluid resolution and symptom
relief17. Successful drainage of biloma with biloma-gastrostomy
has also been reported by Nayak et al.
Consent
Written informed consent was obtained from the patient agreeing
that this manuscript can be published.
Figure 2. Follow-up ultrasound findings. (a) Follow up sonogram
obtained after percutaneous transhepatic cholangiography (images
not retrieved) show a large sonolucent sub hepatic left liver lobe mass
12×11cm, with a wall thickness of 0.22 cm. (b) Follow-up sonogram
obtained after percutaneous transhepatic cholangiography revealed
biloma mass adjacent to the gall bladder with internal debris.
A surgeon was then consulted 6 months after discharge
(June 2017), the following was noted: patient had persistent
epigastric pain, and persistent biloma, despite serial percutane-
ous aspiration and insertion of a pigtail catheter. The patient had
normal blood indices: hemoglobin, 15.2g/dl; white cell count
range, 4300-6000 cells/mm3; serum glutamic-oxaloacetic
transaminase, 6 U/l (normal range, 0-37 U/l); serum glutamic
pyruvic transaminase, 56 U/l (normal range, 0-40 U/l). Open
surgical drainage was performed under general anesthesia, and a
bigger drain tube left in the biloma cavity. The Biloma drained
from 600 ml to 2 mls within 12 days.
From the 13th day after surgery, the volume of discharging bilious
material progressively increased from 2 ml to a maximum of
1270 ml on day 26 after the operation. On day 30 after the
operation, a re-laparotomy was done and a biloma-gastrostomy
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Data availability
All data underlying the results are available as part of the article and
no additional source data are required.
Competing interests
No competing interests were disclosed.
Grant information
This work was supported by the African Academy of Sciences
through a DELTAS Africa Initiative Grant [DEL-15-011], as part of
the THRiVE-2 initiative.
The funders had no role in study design, data collection and analysis,
decision to publish, or preparation of the manuscript.
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