Accepted: 22 October 2016

DOI: 10.1111/petr.12846

CASE REPORT

Combined liver transplant and pancreaticoduodenectomy for

inflammatory hilar myofibroblastic tumor: Case report and
review of the literature

Jennifer Berumen|Patrick McCarty|Jun Mo|Kimberly Newton|

Timothy Fairbanks|Kristin Mekeel|Alan Hemming

University of California San Diego Surgery,

La Jolla, CA, USA Abstract
IMT, previously known as IPT, is a relatively rare tumor that was originally described
Correspondence
Jennifer Berumen, University of California San in the lungs, but case reports have reported the tumor in almost every organ system.
Diego Surgery, La Jolla, CA, USA. Surgical resection is typically the mainstay of therapy; however, tumors have also
Email: jberumen@ucsd.edu
been shown to respond to chemotherapy or anti-inflammatory therapy and some
have spontaneously regressed. We present a literature review and case report rep-
resenting the first documentation to date of liver transplant combined with PD for
surgical resection of a myofibroblastic tumor non-responsive to medical therapy.

KEYWORDS:inflammatory pseudotumor, liver transplantation, myofibroblastic tumor,

pancreaticoduodenectomy

alkaline phosphatase of 1306U/L. Initial MRI of his abdomen dem-

1| INTRODUCTION
onstrated an infiltrative mass lesion centered at the confluence of
the left and right biliary tree encasing the common hepatic artery
IMT, previously known as IPT, is a relatively rare tumor that was origi-
and extending toward the pancreatic head, with significant obstruc-
nally described in the lungs, but case reports have reported the tumor
tive biliary ductal dilatation (Figure1). The patient underwent bilateral
in almost every organ system. Surgical resection is typically the main-
transhepatic percutaneous catheter placement and cholangiogram
stay of therapy; however, tumors have also been shown to respond to
with biliary stricture dilatation. Tumor markers (AFP, CEP, CA 19-9)
chemotherapy or anti-inflammatory therapy and some have sponta-
were normal and biopsy of the mass demonstrated a neutrophilic pre-
neously regressed. For surgical treatment of IMT, either liver transplant
dominance with tissue necrosis without a definite diagnosis, and he
or PD alone has been performed; however, liver transplant combined
was treated for infectious sources with meropenem. A laparoscopic
with PD has never been reported. This case report represents the first
biopsy of the mass showed friable tissue and only demonstrated a re-
documentation to date of liver transplant combined with PD for com-
active lymph node with no evidence of malignancy. Eventually, the bil-
plete surgical resection of a myofibroblastic tumor non-responsive to
iary drains required replacement due to repeated obstruction, and at
medical therapy.
that time, he underwent a repeat CT-guided biopsy of the mass. The
biopsy was consistent with an IMT and stained positive for ALK-1.
After multidisciplinary discussions, chemotherapy for his IMT
2| CASE REPORT
was recommended, and the patient was started on crizotinib, an
ALK inhibitor.1 Surgery was initially deferred due to the apparent
A 13-year-old boy with no significant past medical history initially
involvement of the tumor into the bilateral second-order branches
presented to the emergency room for evaluation of fatigue and pain-
of the biliary tree on imaging along with the apparent encasement of
less jaundice. Laboratory work revealed a bilirubin of 11mg/dL and
the hepatic artery and portal vein. After a month of crizotinib ther-
Abbreviations: ALK, anaplastic lymphoma kinase; ALK-1, anaplastic lymphoma kinase 1; IMT, apy, he developed presumed hepatotoxicity with increasing AST,
inflammatory myofibroblastic tumor; IPT, inflammatory pseudotumor; IVC, inferior vena cava;
ALT, and INR. Repeat imaging demonstrated interval progression in
PD, pancreaticoduodenectomy; RANBP2, ran-binding protein 2; SMV, superior mesenteric
vein; SV, splenic vein. activity of both size and FDG of the hepatic mass without evidence

Pediatric Transplantation 2016; 16 wileyonlinelibrary.com/journal/petr 2016 John Wiley & Sons A/S. | 1
Published by John Wiley & Sons Ltd
 |
2 BERUMEN etal.
R Ten months after his initial presentation, and 4months after his
L attempted resection and listing for liver transplant a donor became
GB
F I G U R E 1 Initial MR cholangiogram 3D sagittal view shows
significant bilateral biliary dilatation and cystic duct dilatation with
obstruction of the common bile duct by the mass. The arrow points
to the area of obstruction. R and L indicate the right and left biliary
systems, and GB indicates the gall bladder
of untreated obstruction (Figure2). With the failure of crizotinib to
decrease the size of the mass and hepatotoxicity, crizotinib was dis-
continued, and he was started on celecoxib therapy in an attempt
to decrease the tumor size. Surgical excision was re-considered
and attempted, with the understanding that liver transplant may be
necessary to remove the tumor if hepatic resection could not be
completed.
At the time of the initial operation, the tumor was found to involve
the bifurcation of the hepatic artery and bifurcation of the hepatic duct
with possible extension into the duodenum; the mass was deemed un-
resectable, and resection was aborted after complete staging, includ-
ing lymph node assessment. With no evidence of metastatic disease,
the patient was listed for liver transplant as the only option for com-
plete resection due to the involvement of the bilateral biliary tree and
encasement of the entire hepatic hilum by tumor. Over the next few
months, he continued on celecoxib therapy while awaiting liver trans-
plant. The patient continued to have recurrent episodes of bacterial
cholangitis and failure to thrive with weight loss but a normal MELD
score of 8 and no evidence of underlying liver disease. MELD excep-
tion points of 30 and then eventually 40 were granted by the UNOS
region 5 regional review board because of the recurrent cholangitis
and the development of antibiotic resistant stenotrophomonas bacte-
ria in his biliary tree.
available. The donor was a healthy 39-year-old woman who died from
trauma and was slightly smaller than our recipient. He was taken to the
operating room for liver transplant. Cold ischemia time was 7hours.
At the time of transplant, the tumor had obviously increased in size
with involvement of the duodenum, head of the pancreas, and portal
vein to the level of the SV/SMV confluence. No lymph node sampling
was attempted as the mass was bulky and inflammatory, making mo-
bilization for sampling difficult without committing to transplantation.
Borders were identified to confirm the feasibility of the anticipated
en bloc resection. The en bloc resection included hepatectomy and
pancreaticoduodenectomy to resect the involved portion of pancreas
and duodenum, and the portal vein was removed to the level of the
SV/SMV confluence. The medial portion of the IVC was inseparable
from the mass; therefore, the IVC was included in the specimen and
the donor liver anastomosed with a bi-caval technique. The hepatic
artery was completely encased by tumor, so the resection also in-
cluded the common hepatic artery down to the level of the celiac axis
(Figure3). With the extended resection of the portal vein and hepatic
artery along with the specimen, additional vascular reconstruction was
necessary. The donor portal vein was anastomosed to the SV/SMV
confluence via a donor iliac vein jump graft. The donor celiac axis was
anastomosed to a donor iliac artery jump graft off the patients infra-
renal aorta (Figure4). Two separate roux limbs were fashioned for the
choledochojejunostomy and pancreaticojejunostomy anastomoses,
and a gastrojejunostomy was created. Two separate enteric limbs were
used to separately drain pancreatic and biliary secretions to decrease
the chance of catastrophic complications if a leak occurred from either
the biliary, or in particular, pancreatic anastomosis. In addition, if a re-
transplant or further tumor resection is required, the presence of two
roux limbs will facilitate a safer reconstruction. The reconstructions
are shown in Figure5.
Postoperatively, the patient was maintained on intravenous hep-
arin with a target PTT of 50-60 and then transitioned to enoxaparin
for 1month. No further anticoagulation or specific antiplatelet agents
were used, but he continued on celecoxib. With no evidence of ac-
tive infection at the time of transplant, additional antibiotics were
F I G U R E 2 CT scan prior to attempted
resection shows a large ill-defined mass
with narrowing of the portal vein (black
arrow) and encasement of the hepatic
artery (white arrow). Biliary drains are
visible, and rounded lines mark the
presumed tumor margins
BERUMEN etal.
Duodenum Antrum
Head of pancreas
Right lobe liver
F I G U R E 3 Complete en bloc resection including hepatectomy
and pancreaticoduodenectomy specimen. Tumor was encasing the
complete hepatic hilum
IV
IA
F I G U R E 4 Portal vein reconstruction with iliac vein jump graft (IV)
and iliac artery aortic jump graft (IA). Feeding tube is in the pancreatic
duct (arrow)
not given for his history of cholangitis apart from 7days of periop-
erative meropenem. He had a relatively uneventful postoperative
course with no delayed gastric emptying or other surgical issues and
was discharged home 2weeks post-transplant. As an outpatient, the
patient did have mildly increased stool fat content and was briefly on
pancreatic enzymes until this resolved, but there was no difficulty
with blood glucose management or immunosuppression absorption.
Immunosuppression consisted of steroid induction with standard
triple therapy maintenance of prednisone, mycophenolate, and tac-
rolimus with a goal level of 10-12ng/mL. Steroids were weaned off
at 9months, and he remains on dual therapy with no episodes of
rejection.
Pathology demonstrated an 8cm ALK-1-positive myofibroblas-
tic tumor with extension into the head of the pancreas, duodenum,
and bilateral lobes of the liver (Figure6). The large vessels were
completely encased by tumor, but there was no definite vascular in-
vasion or lymph node involvement. The hilar biliary tree was involved
|
3
with the mass. One year later, he remains well with excellent liver
function and is thriving. He has gained weight and is participating in
sports and activities with a full energy level. His current follow-up
plan is contrast enhanced MRIs every 3months, and a PET scan
every 6months until he is 3years post-transplant, at which point the
imaging frequency will decrease. His latest studies show no evidence
of recurrence.
3|REVIEW/DISCUSSION
IMT has previously been included in a group known as IPT, which
have also been described as plasma cell granulomas, histiocytomas,
fibroxanthomas, and inflammatory fibrosarcomas.24 More recent
descriptions have histologically described IMTs as different from the
general group of IPTs with their own biology and behavior, but many
past reports include them all within the same category, making data
interpretation difficult.5 IMTs are by themselves considered to have
intermediate biological potential by the World Health Organization.6,7
The most common presentation is in the lung, followed by the abdo-
men and retroperitoneum, but reports have described tumors in al-
most every organ system including extremity disease, head and neck
involvement, retroperitoneal laryngeal, ovarian, tracheal, esophageal,
renal, and splenic tumors.712 The typical age of presentation is in
children and young adults, but can occur at any age. The mechanism
of tumor development is unknown, but theories include response to
tissue injury, infection, or an autoimmune process8,13; metastasis is
uncommon.
Clinically, IMTs may present as slow growing masses, and symp-
toms are dependent on location and involved structures but usually
related to obstructive mass symptoms. Our patient presented with bil-
iary obstruction due to the hilar location. Imaging may not be helpful
with the diagnosis, as some may appear as amorphous masses with
calcifications on x-ray or a solid well-defined mass on CT. MRI also
has variable findings, and the tumor may appear as hypo-intense on
T1 or hyperintense on T2 imaging, but there are no standard defined
radiologic characteristics.4,14,15 Further workup is usually necessary
with biopsy and staining to confirm the diagnosis if the clinical picture
is suspicious for IMT.
Histopathologically, IMTs appear as a bland spindle cell prolifer-
ation with a range of inflammatory cells, primarily plasma cells, and
lymphocytes, but also containing eosinophils and neutrophils. Some
may contain calcifications, hemorrhage, or necrosis. Three basic pat-
terns have been described: a myxoid/vascular pattern with a fasciitis-
like appearance and loosely arranged spindle cells, a compact spindle
cell pattern with a collagen background, and a hypocellular fibrosis pat-
tern that has fewer cells overall with elongated spindle cells in dense
collagen. Some also contain foamy histiocytes and plasma cells, with
the combination of these patterns creating a broad range of potential
findings.6,11 Some tumors exhibit chromosomal changes, and those
changes can definitively diagnose IMTs.12
ALK-1 positivity is present with diffuse staining in about 50% of
tumors. ALK expression indicates a rearrangement of the ALK locus
 |
4 BERUMEN etal.

PV

(A) (B)
IV

SMV

CA

(C)
SMA
CA

PV

IA
SV
Pancreas IVC Aorta

IVC

(D)

SMV
GJ

PJ
CJ

F I G U R E 5 Schematic of the reconstructions performed. PV, donor portal vein; CA, celiac axis; SV, splenic vein; SMV, superior mesenteric
vein; IV, iliac vein graft; IA, iliac artery jump graft; CJ, choledochojejunostomy; PJ, pancreaticojejunostomy; GJ, gastrojejunostomy. A, After
completion of the bi-caval anastomosis of the liver transplant, the major hepatic vessels required jump grafts to complete anastomoses off
tension. B, Completion of the portal venous anastomosis. An iliac vein jump graft was used to connect between the confluence of the SMV
and SV and the donor PV. C, The arterial connection was completed with an infrarenal aortic jump graft to the donor celiac axis using an
iliac artery jump graft (IA). D, The bowel was reconnected using two separate roux limbs to create the choledochojejunostomy (CJ) and the
pancreaticojejunostomy (PJ) followed by a gastrojejunostomy (GJ)

on chromosome 2p23 that encodes a tyrosine kinase oncogene with
multiple fusion partners.1 ALK-1 positivity does not appear to be as-
sociated with any local aggression or specific tumor behavior patterns,
but some studies have shown IMTs without ALK-1 expression appear
more likely to develop metastatic disease. Another genetic variant,
RANBP2, may also be associated with a more aggressive tumor and
16
the development of recurrence and metastatic disease. Along those
lines, larger tumors also appear more likely to have local recurrence.17
Recently, another variant of IMT has been described called epithelioid
inflammatory myofibroblastic sarcoma, which has ALK expression in
a nuclear membrane or a peri-nuclear staining pattern histologically.
These tumors are much more aggressive and have a high propensity
for local recurrence and metastasis.18 Overall, the behavior of the
tumor and response to treatment are not consistently predictable by
any specific histological findings.12 Crizotonib is a newer drug used
with anti-ALK activity that has been shown to decrease tumor size in
ALK positive tumors.1,19 Crizotonib was used in our patient, but did
not decrease the size of his tumor.

F I G U R E 6 A moderately cellular
infiltrating tumor consists of plump
fibroblastic spindle cells with abundant
inflammatory cells (A, B) including many
plasma cells (C). The tumor cells show
positive ALK-1 immunohistochemical stain
(D). Tumor infiltrates and replaces a large
portion of liver parenchyma (A, B) and
partially involves pancreatic head (E) and
duodenal wall (F). Arrows indicate residual
bile ducts (B), plasma cells (C), ALK-1-
positive tumor cells (D), residual pancreatic
tissue (E), and duodenal glands (F)
BERUMEN etal.
Although IPTs in the literature have been reported to spon-
taneously regress or improve with steroid therapy, 14,2024
the
interchangeable use of IPT and IMT in the literature makes data in-
terpretation regarding IMTs difficult. Successful treatment and tumor
regression have been reported with many methods, including corti-
costeroids, anti-inflammatories, antibiotics, chemotherapy, radiation,
and surgery. However, tumors definitively reported as IMT by histo-
pathology, and not IPT, are locally progressive and appear to need
surgical resection, especially with failure of medical therapy.5,9,12
Surgical resection with negative margins remains the standard of
therapy for IMTs if it is technically feasible. Long-term prognosis is
difficult to determine, but seems good if there is a response to med-
ical therapy, or if complete surgical excision is possible. Tumors that
are larger (>10cm), not ALK positive, and that are in difficult to excise
areas like the retroperitoneum have a higher rates of recurrence, even
up to 25% in the literature. Only approximately 5% of the tumors typ-
ically develop distant metastasis; however, the RANBP2 tumors have
shown recurrence rates of up to 88% with metastasis rates of 25%.16
There have been previous reports of hilar IMTs,2533 and several
liver or hilar IMTs have been treated with liver transplant.25,27,29,33
IMTs also have been treated in previous reports with pancreaticodu-
odenectomy with good results and low recurrence rates.3436 One
report performing pancreatic resection and transverse colectomy for
an IMT also required reconstruction of the mesenteric vasculature,9
and another required multivisceral transplantation including liver and
pancreas.37 Liver transplant combined with PD has been reported for
other indications generally in adults,3843 typically in the treatment
of cholangiocarcinoma, but not for an IMT. This is the first reported
case of combined liver transplant and pancreaticoduodenectomy with
vascular reconstruction for IMT. Patients receiving combined liver
transplant and PD for other diseases seem to have few immediate
complications related to the procedure, and most of the morbidity
and mortality are related to tumor recurrence and sepsis further on
in their course,38 indicating the simultaneous combination is a viable
treatment option. With IMTs, the need for complete surgical excision
of these tumors may require innovative strategies. This tumor was
particularly difficult to excise without a dramatic resection given its dif-
fuse involvement of the liver hilum, head of pancreas, and associated
vascular structures.
With current follow-up at 1year after liver transplant and PD, our
patient has had no evidence of recurrent disease and has returned
to normal life and school activities. Longer-term follow-up is required
prior to claiming complete success; however, the current result is
promising.
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How to cite this article: Berumen, J., McCarty, P., Mo, J., Newton, K.,
Fairbanks, T., Mekeel, K. and Hemming, A. (2016), Combined liver
transplant and pancreaticoduodenectomy for inflammatory hilar
myofibroblastic tumor: Case report and review of the literature.
Pediatric Transplantation, 00: 16. doi: 10.1111/petr.12846