A Hashimoto

Adrian Waugh

AP Burke

B Kumar

Bjoern Kitzing

DE Fish

E Gorgun

EC Ebert

HK Ha

J Juliano

Jane Clayton

JC Hoffmann

Kevin C Allman

KT Chen

Neil McGill

P Rajvanshi

S Manoharan

Sandra O'Toole

SR Fernandes

TR Cupps

English

PubMed

Crossref

Hepatobiliary scintigraphy in vasculitis of the gallbladder as a manifestation of polyarteritis nodosa: a case report

Springer - Publisher Connector

BioMed CentralCases Journal
ssOpen AcceCase Report
Hepatobiliary scintigraphy in vasculitis of the gallbladder as a 
manifestation of polyarteritis nodosa: a case report
Bjoern Kitzing*1, Sandra O'Toole2, Adrian Waugh1, Jane Clayton3, 
Neil McGill3 and Kevin C Allman1
Address: 1Department of PET and Nuclear Medicine, Royal Prince Alfred Hospital, Missenden Road, Sydney, New South Wales, Australia, 
2Department of Anatomical Pathology, Royal Prince Alfred Hospital, Missenden Road, Sydney, New South Wales, Australia and 3Department of 
Rheumatology, Royal Prince Alfred Hospital, Missenden Road, Sydney, New South Wales, Australia
Email: Bjoern Kitzing* - bkitzing@gmail.com; Sandra O'Toole - sot@yahoo.com; Adrian Waugh - awaugh@gmail.com; 
Jane Clayton - jane_clayton@hotmail.com; Neil McGill - neil_mcgill@gmail.com; Kevin C Allman - allman@yahoo.com
* Corresponding author    
Abstract
Introduction: Polyarteritis nodosa can on rare occasions manifest itself as vasculitis of the
gallbladder. Patients typically present with right upper quadrant pain and are initially worked up for
cholecystitis. The definitive diagnosis is then usually based on surgical and histopathological findings.
Case presentation: In this case a 23-year-old Caucasian female presented with a 3 week history
of right upper quadrant pain and fevers.
Conclusion: The clinical pathway and imaging findings of a rare case of gallbladder vasculitis as a
manifestation of polyarteritis nodosa are demonstrated.
Introduction
We present the clinical pathway of a rare case of gallblad-
der vasculitis secondary to polyarteritis nodosa. The clini-
cal pathway demonstrates the importance of the clinical
presentation and the use of medical imaging in confirm-
ing the diagnosis.
Case presentation
A 23-year-old Caucasian female presented with a 3 week
history of right upper quadrant pain, fevers, bilateral
ankle pain and swelling with an accompanying rash. Her
past medical history consisted of a previous hospital
admission 3 months earlier due to an acute inflammatory
polyarthritis associated with fever. During that admission
the patient had developed a maculopapular rash over the
trunk and limbs. Skin biopsies of the right thigh were
taken which showed leukocytoclastic vasculitis. Symp-
toms improved after commencement of prednisone and
the patient was discharged.
When the patient represented she had an elevated C-reac-
tive protein 168 mg/L (normal: <12), erythrocyte sedi-
mentation rate 76 mm/hr (0-12) and alkaline
phosphatase 162 U/L (30-130). All her other blood
results were normal. Rheumatoid Factor, ANCA and HLA
B27 were negative as were blood cultures and viral serolo-
gies. Hepatobiliary scintigraphy was requested to investi-
gate hepatobiliary function (Figures 1, 2). The study was
performed with 220 MBq (5.9 mCi) Tc-99m disofenin.
The vascular phase appeared normal. The parenchymal
phase showed an enlarged left lobe of the liver. Bile pro-
duction was normal with rapid appearance in the intrahe-
Published: 10 December 2009
Cases Journal 2009, 2:9300 doi:10.1186/1757-1626-2-9300
Received: 30 November 2009
Accepted: 10 December 2009
This article is available from: http://www.casesjournal.com/content/2/1/9300
© 2009 Kitzing et al; licensee BioMed Central Ltd. 
This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), 
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.Page 1 of 4
(page number not for citation purposes)
Cases Journal 2009, 2:9300 http://www.casesjournal.com/content/2/1/9300patic ducts and normal passage of bile into the bowel.
However, the gallbladder filled slowly during the study.
An outpatient computed tomography scan which had
been performed prior to admission showed a thickened
gallbladder wall with surrounding fluid but no evidence
of stones or duct dilatation (Figure 3).
Based on these findings and the clinical picture a laparo-
scopic cholecystectomy was performed. The postoperative
recovery was unremarkable and the patient was dis-
charged two days later and subsequently had no further
right upper quadrant pain. Clinical follow-up one month
later showed that she was still free of abdominal pain but
had developed symptoms consistent with mononeuritis
multiplex. Polyarteritis nodosa was considered the most
likely diagnosis clinically.
Macroscopic examination of the gallbladder showed no
gross abnormality. However, microscopic examination
revealed a striking vasculitis affecting small to medium
sized vessels, predominantly arteries. There was fibrinoid
necrosis of many vessels associated with an inflammatory
cell infiltrate, composed predominantly of lymphocytes
with very occasional eosinophils (Figure 4). Almost every
vessel in the histological sections was affected and some
showed thrombotic occlusion. The findings were consist-
ent with polyarteritis nodosa.
Conclusion
Polyarteritis nodosa (PAN) is an uncommon multisys-
tem, necrotizing vasculitis of small- and medium-sized
muscular arteries in which involvement of renal and vis-
ceral arteries is characteristic [1]. It was first described in
its classic form by Kussmaul in 1866. The diagnosis of
PAN is based on the demonstration of specific signs of
vasculitis on biopsy material of involved organs as there
are no diagnostic serologic tests. We present the hepato-
biliary scintigraphy findings of a patient with cholecystitis
which was a rare clinical manifestation of PAN.
Vasculitis is a clinicopathologic process characterized by
inflammation of and damage to blood vessels. Polyarteri-
tis nodosa (PAN) can cause specific complaints related to
the vascular involvement within a particular organ system
which may dominate the presenting clinical picture as
well as the entire course of the illness. Involvement of the
gastrointestinal tract has a 44% incidence in patients with
classic PAN. Clinical manifestations include abdominal
pain, nausea and vomiting, bleeding, bowel infarction
and perforation, cholecystitis, as well as hepatic and pan-
creatic infarction. At autopsy 10% of patients with classic
PAN have involvement of the gallbladder.
Hepatobiliary scintigraphy of a 23-year-old white female with a three week history of right upper quadrant pain after injec-tion of 220 MBq (5.9 mC ) Tc-99m disofeniFigur  1
Hepatobiliary scintigraphy of a 23-year-old white 
female with a three week history of right upper quad-
rant pain after injection of 220 MBq (5.9 mCi) Tc-
99m disofenin. Anterior dynamic views of hepatobiliary 
scintigraphy showing normal bile production with rapid 
appearance in the intrahepatic ducts and normal passage of 
bile into the bowel. A very small amount of activity entered 
the gallbladder early in the study (arrow).
Hepatobiliary scintigraphy of a 23-year-old white female with a three week history of right upper quadrant pain after injec-tion of 220 MBq (5.9 mC ) Tc-99m disofeninFigur  2
Hepatobiliary scintigraphy of a 23-year-old white 
female with a three week history of right upper quad-
rant pain after injection of 220 MBq (5.9 mCi) Tc-
99m disofenin. Anterior delayed static view of hepatobil-
iary scintigraphy demonstrating progressive build up of activ-
ity in the gallbladder (arrow) after 57.5 minutes.Page 2 of 4
(page number not for citation purposes)
Cases Journal 2009, 2:9300 http://www.casesjournal.com/content/2/1/9300Gallbladder vasculitis of varying aetiology is rare but has
been well described in the medical literature [2-13].
Abdominal pain, especially in the right upper quadrant is
the most common presenting symptom. Liver function
tests may be abnormal but as patients are usually thought
to have cholecystitis secondary to cholelithiasis abdomi-
nal ultrasound imaging is performed sometimes followed
by computed tomography [14]. Following laparoscopic or
open cholecystectomy the diagnosis is then made based
on the intraoperative and histological evidence.
In this reported case the hepatobiliary scintigraphy find-
ings were consistent with slow filling of the gallbladder
most likely due to oedematous swelling of the vasculitic
tissue as was subsequently demonstrated by the histopa-
thology. Appropriate knowledge and awareness of gall-
bladder vasculitis secondary to polyarteritis nodosa will
help interpret non-specific yet clearly abnormal hepato-
biliary scintigraphy findings.
Abbreviations
ANCA: anti-neutrophil cytoplasmic antibody; HLA:
human leukocyte antigen; PAN: polyarteritis nodosa;
IDA: iminodiacetic acid.
Consent
Written informed consent was obtained from the patient
for publication of this case report and all accompanying
images. A copy of the written consent is available for
review by the Editor-in-Chief of this journal.
Competing interests
The authors declare that they have no competing interests.
Authors' contributions
BK, SOT and AW made substantial contributions to con-
ception and design and drafted the manuscript. JC, NM
and KCA revised it critically for important intellectual
content and gave final approval of the version to be pub-
lished.
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Hepatobiliary scintigraphy in vasculitis of the gallbladder as a manifestation of polyarteritis nodosa: a case report

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