DOI:10.2214/AJR.07.3663
AJR 2008; 191:1255-1262
© American Roentgen Ray Society
Inflammatory Pseudotumor (Myoblastic Tumor) of the Genitourinary Tract
Sung Bin Park1,2,
Kyoung-Sik Cho2,
Jeong Kon Kim2,
Jong Hwa Lee1,
Ae Kyung Jeong1,
Woon Jung Kwon1 and
Hak Hee Kim2
1 Department of Radiology, Ulsan University Hospital, University of Ulsan, 290-3
Jeonha-dong, Ulsan, 682-714, Korea.
2 Department of Radiology and Research Institute of Radiology, Asan Medical
Center, University of Ulsan, Seoul, Korea.
Received January 10, 2008;
accepted after revision April 22, 2008.
Address correspondence to S. B. Park
(pksungbin{at}paran.com).
Abstract
OBJECTIVE. The purpose of this article is to describe imaging
features of the following genitourinary tract locations that can involve
inflammatory pseudotumor: kidney, adrenal gland, retroperitoneum or pelvis,
bladder, and other rare locations.
CONCLUSION. Although definite radiologic differentiation from
malignancy is not clearly possible, we suggest that familiarity with the
manifestations of inflammatory pseudotumor can help avoid unnecessary radical
surgery before histopathologic proof of malignancy is obtained.
Keywords: CT • genitourinary tract • inflammatory myofibroblastic tumor • inflammatory pseudotumor
Introduction
Inflammatory pseudotumor is a rare benign condition of unknown cause. It is
characterized by the presence of a mass that may mimic malignancy and is
composed of spindle cells mixed with variable amounts of extracellular
collagen, lymphocytes, and plasma cells. The imaging characteristics and
locations of inflammatory pseudotumor in the genitourinary tract vary widely,
with mostly large masses mimicking malignant lesions. Inflammatory pseudotumor
can also manifest as multifocal lesions such as lymphoma and local tumor
recurrence or metastasis. Although the imaging features of inflammatory
pseudotumor are nonspecific and not characteristic, the role of the
radiologist is to ensure that inflammatory pseudotumor is a diagnosis that is
suggested preoperatively and differentiated from malignant lesions by either
particular imaging findings or imaging-guided biopsy.
In this article, we describe the imag ing features of the following
inflammatory pseudotumors that involve the genitourinary tract: kidney,
adrenal gland, retroperitoneum or pelvis, bladder, and other rare
locations.
Pathogenesis and Clinical Manifestations of Inflammatory Pseudotumor
Inflammatory pseudotumor is a chronic inflammatory tumefaction of unknown
origin. Because the term "inflammatory pseudotumor" is nonspecific
and the lesions have a variety of histologic presentations, several
alternative names have been used: inflammatory myofibroblastic tumor, plasma
cell granuloma or pseudotumor, xanthomatous pseudo tumor, pseudosarcomatous
myofibroblastic pro liferation, inflammatory myofibroblastic proliferation,
and myofibroblastoma.
The cause and pathogenesis of inflammatory pseudotumor still remain
controversial. Inflammatory pseudotumor is characterized histologically by the
presence of acute and chronic inflammatory cells with a variable fibrous
response. The microscopic features of inflammatory pseudotumor are dominated
by differentiated vimentin-positive myofibroblastic spindle cells, a variably
prominent collagenous stroma, and an inflammatory component consisting mainly
of lymphocytes and plasma cells.
Recently, the term "inflammatory myofibroblastic tumor" has
come to be commonly used on the basis of electron microscopic and
immunohistochemical findings
[1,
2]. The World Health
Organization continues to classify inflammatory myofibroblastic tumor as a
distinct borderline lesion with uncertainty as to whether it is reactive or
neoplastic in nature. Recent studies have revealed cytogenetic clonal
abnormality and anaplastic lymphoma kinase expression, suggesting a neoplastic
cause [3,
4].
Inflammatory pseudotumor most commonly involves the lung and orbit, but it
has been described in almost any location, in both sexes, and at all ages
[1]. Most cases occur in
childhood or early adulthood and may be asymptomatic or associated with
constitutional complaints or symptoms including unexplained fever or weight
loss and related to local mass effects
[2,
5]. Inflammatory pseudo tumor
includes a wide spectrum of biologic behavior, varying from the more frequent
benign lesions to the rare tumors that are multifocal and prone to recurrence.
Inflammatory pseudotumors clinically masquerade as malignant growths, and
their clini cal significance thus lies in the difficulty encountered in
preoperatively excluding malignancy. The preoperative recognition of this
benign lesion by imaging-guided biopsy is important to avoid unnecessarily
radical resection. Although several cases of spontaneous regression and
response to medical treatment have been reported, in consideration of its
pathologic features and clinical manifestations, complete surgical resection
is mandatory. The prognosis of inflammatory pseudotumor is generally
considered to be favorable if complete surgical resection is performed, with
only a rare incidence of malignant transformation and remote metastasis
[6].
Radiologic Features of Inflammatory Pseudotumor
Although the radiologic findings of inflammatory pseudotumor are
nonspecific, particular findings are observed. Sonographic findings show a
variable pattern of echogenicity, and the lesion has been described as hypoor
hyperechogenic with ill-defined or well-defined margins
[7]. Prominent vascularity may
be shown with color or power Doppler sonography, especially contrast-enhanced
power Doppler sonography. Contrast-enhanced CT may show homo- or heterogeneity
and hypo-, iso-, or hyperdensity
[8]. MRI shows a hypointense
lesion on T1- and T2-weighted images (possibly reflecting the fibrotic change)
and shows marked gadolinium enhancement. In these gadolinium-enhanced images
of inflammatory pseudotumor, delayed enhancement has frequently been observed,
probably because of the accumulation of extravascular contrast media in the
fibrotic component within the mass
[9]. These variable radiologic
findings may be attributed to varying degrees of fibrosis, cellular
infiltration, and dynamic change occurring during the inflammatory
process.
Inflammatory Pseudotumor of the Kidney
Renal inflammatory pseudotumor is very rare. Cases have been reported in
patients between the ages of 8 and 68 years, most of them male. Patients with
renal inflammatory pseudotumor usually present with flank pain and hematuria.
Laboratory abnormalities have not been reported except microscopic hematuria.
One reported case was detected incidentally
[10].
Various radiologic findings of renal inflammatory pseudotumor can be shown.
Renal inflammatory pseudotumor can be seen as a hypo- or heterogeneous echoic
mass on sonography, well-defined hypoechoic mass with intratumoral vascularity
on enhanced power Doppler sonography, low-attenuation mass on CT (Figs.
1A,
1B,
1C and
2A,
2B), and hypovascular lesion on
MRI [10].
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Fig. 1A —67-year-old man with inflammatory pseudotumor of kidney.
Unenhanced (A), corticomedullary phase (B), and early excretory
phase (C) images of dynamic CT of kidney show solid mass in lower pole
of left kidney. Subtle increased density on unenhanced scan may indicate
calcification. On corticomedullary and early excretory phases, mass enhanced
and washed out (arrows, B and C). Primary radiologic
diagnosis was renal cell carcinoma. Left radical nephrectomy was performed.
Pathologic results indicated inflammatory pseudotumor.
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Fig. 1B —67-year-old man with inflammatory pseudotumor of kidney.
Unenhanced (A), corticomedullary phase (B), and early excretory
phase (C) images of dynamic CT of kidney show solid mass in lower pole
of left kidney. Subtle increased density on unenhanced scan may indicate
calcification. On corticomedullary and early excretory phases, mass enhanced
and washed out (arrows, B and C). Primary radiologic
diagnosis was renal cell carcinoma. Left radical nephrectomy was performed.
Pathologic results indicated inflammatory pseudotumor.
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Fig. 1C —67-year-old man with inflammatory pseudotumor of kidney.
Unenhanced (A), corticomedullary phase (B), and early excretory
phase (C) images of dynamic CT of kidney show solid mass in lower pole
of left kidney. Subtle increased density on unenhanced scan may indicate
calcification. On corticomedullary and early excretory phases, mass enhanced
and washed out (arrows, B and C). Primary radiologic
diagnosis was renal cell carcinoma. Left radical nephrectomy was performed.
Pathologic results indicated inflammatory pseudotumor.
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Fig. 2A —46-year-old man with inflammatory pseudotumor of pancreas and
kidney. Inflammatory pseudotumor manifests as multifocal disease such as
lymphoma. Contrast-enhanced CT scans show bulbous enlargement of the pancreas
(arrow, A) and multifocal low-attenuation lesions
(arrowheads, A) in both kidneys. Multiple enlarged nodes in
porta hepatis and portocaval space were also present (not shown). Primary
radiologic diagnosis was lymphoma. Sonography-guided kidney biopsy was
performed. Pathologic results indicated inflammatory pseudotumor.
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Fig. 2B —46-year-old man with inflammatory pseudotumor of pancreas and
kidney. Inflammatory pseudotumor manifests as multifocal disease such as
lymphoma. Contrast-enhanced CT scans show bulbous enlargement of the pancreas
(arrow, A) and multifocal low-attenuation lesions
(arrowheads, A) in both kidneys. Multiple enlarged nodes in
porta hepatis and portocaval space were also present (not shown). Primary
radiologic diagnosis was lymphoma. Sonography-guided kidney biopsy was
performed. Pathologic results indicated inflammatory pseudotumor.
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Inflammatory Pseudotumor of the Adrenal Gland
Endocrine organ involvement by inflammatory pseudotumor including the
adrenal gland is very rare. Adrenal inflammatory pseudotumor may be seen as a
nonspecific adrenal solid mass (Fig.
3A,
3B,
3C,
3D) and patients may suffer
from endocrine symptoms such as amenorrhea
[11].
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Fig. 3A —38-year-old man with inflammatory pseudotumor of adrenal
gland. Inflammatory pseudotumor manifests as local tumor recurrence and
metastasis on follow-up images. Contrast-enhanced CT scan shows
heterogeneously enhancing mass (arrow) in right adrenal gland.
Perihepatic fluid collection is also evident.
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Fig. 3B —38-year-old man with inflammatory pseudotumor of adrenal
gland. Inflammatory pseudotumor manifests as local tumor recurrence and
metastasis on follow-up images. Axial T2-weighted MR image (B) shows
heterogeneous signal intensity mass in right adrenal gland. Contrast-enhanced
axial T1-weighted MR image (C) shows heterogeneous and peripheral
enhancement. Primary radiologic diagnosis was primary adrenal tumor. Right
adrenalectomy was performed. Pathologic results indicated inflammatory
pseudotumor.
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Fig. 3C —38-year-old man with inflammatory pseudotumor of adrenal
gland. Inflammatory pseudotumor manifests as local tumor recurrence and
metastasis on follow-up images. Axial T2-weighted MR image (B) shows
heterogeneous signal intensity mass in right adrenal gland. Contrast-enhanced
axial T1-weighted MR image (C) shows heterogeneous and peripheral
enhancement. Primary radiologic diagnosis was primary adrenal tumor. Right
adrenalectomy was performed. Pathologic results indicated inflammatory
pseudotumor.
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Fig. 3D —38-year-old man with inflammatory pseudotumor of adrenal
gland. Inflammatory pseudotumor manifests as local tumor recurrence and
metastasis on follow-up images. At 2-year follow-up, contrast-enhanced CT scan
shows heterogeneously enhancing large mass in right adrenalectomy site,
presumed to be local tumor recurrence. Chest CT scan also showed multiple
metastatic pulmonary nodules (not shown).
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Inflammatory Pseudotumor of the Retroperitoneum and Pelvis
Inflammatory pseudotumor of the retroperitoneum and pelvis shows a large
softtissue mass on imaging studies (Fig.
4A,
4B,
4C,
4D). As in other sites, these
may often mimic malignancy such as rhabdomyosarcoma (Fig.
5A,
5B). A large infiltrative
lesion exhibiting significant mass effect on neighboring structure is present
[12,
13].
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Fig. 4A —47-year-old woman with inflammatory pseudotumor of
retroperitoneum. Contrast-enhanced CT scans show homogeneously enhancing mass
(arrow, A) in aortic bifurcation area. Nonvisualization of
left iliac vein, probably due to thrombus, is evident.
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Fig. 4B —47-year-old woman with inflammatory pseudotumor of
retroperitoneum. Contrast-enhanced CT scans show homogeneously enhancing mass
(arrow, A) in aortic bifurcation area. Nonvisualization of
left iliac vein, probably due to thrombus, is evident.
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Fig. 4C —47-year-old woman with inflammatory pseudotumor of
retroperitoneum. Axial T2-weighted MR image (C) shows slightly
high-signal-intensity mass in retroperitoneum. Contrast-enhanced axial
T1-weighted MR image (D) shows peripheral rim enhancement. Primary
radiologic diagnosis was retroperitoneal sarcoma with associated thrombosis.
Results of surgical resection indicated inflammatory pseudotumor.
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Fig. 4D —47-year-old woman with inflammatory pseudotumor of
retroperitoneum. Axial T2-weighted MR image (C) shows slightly
high-signal-intensity mass in retroperitoneum. Contrast-enhanced axial
T1-weighted MR image (D) shows peripheral rim enhancement. Primary
radiologic diagnosis was retroperitoneal sarcoma with associated thrombosis.
Results of surgical resection indicated inflammatory pseudotumor.
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Fig. 5A —14-year-old boy with inflammatory pseudotumor of pelvis.
Inflammatory pseudotumor manifests as a large pelvic mass such as
rhabdomyosarcoma. Contrast-enhanced CT scan shows hydronephrosis
(arrow) of left kidney and subcapsular hematoma formation
(arrowhead).
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Fig. 5B —14-year-old boy with inflammatory pseudotumor of pelvis.
Inflammatory pseudotumor manifests as a large pelvic mass such as
rhabdomyosarcoma. Contrast-enhanced CT scan of lower abdomen and pelvis shows
a large heterogeneous enhancing mass (arrow). Primary radiologic
diagnosis was malignant neoplasm such as rhabdomyosarcoma.
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Inflammatory Pseudotumor of the Bladder
Inflammatory pseudotumor of the bladder was first described by Roth
[14] in 1980. The bladder is a
common site of inflammatory pseudotumor in the genitourinary tract. This
entity is rare in children. It can occur at any age but typically appears in
young adults, with an average age of 28 years. About twice as many women as
men have been reported [15].
Painless gross hematuria from exophytic and ulcerated lesions is the most
common initial manifestation and may result in anemia. Other symptoms include
frequency of urination and dysuria, and urinary tract obstruction can also
occur. The lesion may be found at any site in the bladder. However, the
trigone has not been involved except for secondary trigonal invasion from
lesions developing on the posterior wall of the bladder. The tumor appears as
a polypoid intraluminal mass or a submucosal mass with or without extension
into the perivesical fat [16].
Inflammatory pseudotumor should be considered when an enhancing tumor is
surrounded by a clot, particularly in young adults (Fig.
6A,
6B,
6C). A good prognosis can be
expected with this condition if complete resection, especially partial
cystectomy (Fig. 7A,
7B,
7C,
7D,
7E,
7F,
7G,
7H,
7I,
7J) and transurethral
resection is performed.
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Fig. 6A —13-year-old boy with inflammatory pseudotumor of bladder who
complained of painless hematuria. Gray-scale sonography (A) and power
Doppler sonography (B) images show protruded intraluminal mass in
bladder. Increased vascularity (arrow, B) on power Doppler
sonography is evident.
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Fig. 6B —13-year-old boy with inflammatory pseudotumor of bladder who
complained of painless hematuria. Gray-scale sonography (A) and power
Doppler sonography (B) images show protruded intraluminal mass in
bladder. Increased vascularity (arrow, B) on power Doppler
sonography is evident.
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Fig. 6C —13-year-old boy with inflammatory pseudotumor of bladder who
complained of painless hematuria. Contrast-enhanced CT scan shows strongly
enhancing mass (arrow) in bladder. Cystoscopic biopsy was performed.
Pathologic results indicated inflammatory pseudotumor.
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Fig. 7A —21-year-old woman with inflammatory pseudotumor of bladder.
Inflammatory pseudotumor manifests as a large multiloculated pelvic mass such
as ovarian tumor. Contrast-enhanced CT scans show heterogeneous enhancing mass
in pelvis. Normal-appearing left ovary (arrow, C) and no
visible right ovary are evident. So, right ovarian tumor was considered.
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Fig. 7B —21-year-old woman with inflammatory pseudotumor of bladder.
Inflammatory pseudotumor manifests as a large multiloculated pelvic mass such
as ovarian tumor. Contrast-enhanced CT scans show heterogeneous enhancing mass
in pelvis. Normal-appearing left ovary (arrow, C) and no
visible right ovary are evident. So, right ovarian tumor was considered.
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Fig. 7C —21-year-old woman with inflammatory pseudotumor of bladder.
Inflammatory pseudotumor manifests as a large multiloculated pelvic mass such
as ovarian tumor. Contrast-enhanced CT scans show heterogeneous enhancing mass
in pelvis. Normal-appearing left ovary (arrow, C) and no
visible right ovary are evident. So, right ovarian tumor was considered.
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Fig. 7D —21-year-old woman with inflammatory pseudotumor of bladder.
Inflammatory pseudotumor manifests as a large multiloculated pelvic mass such
as ovarian tumor. Axial (D) and coronal (E) T2-weighted MR
images show multiloculated large mass in pelvis with compression to bladder.
Normal-appearing left ovary (arrow, E) and no visible right
ovary are evident.
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Fig. 7E —21-year-old woman with inflammatory pseudotumor of bladder.
Inflammatory pseudotumor manifests as a large multiloculated pelvic mass such
as ovarian tumor. Axial (D) and coronal (E) T2-weighted MR
images show multiloculated large mass in pelvis with compression to bladder.
Normal-appearing left ovary (arrow, E) and no visible right
ovary are evident.
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Fig. 7F —21-year-old woman with inflammatory pseudotumor of bladder.
Inflammatory pseudotumor manifests as a large multiloculated pelvic mass such
as ovarian tumor. Sagittal contrast-enhanced T1-weighted MR image shows
enhancement of septa and compression to bladder. Partial cystectomy was
performed. Pathologic results indicated inflammatory pseudotumor arising from
urinary bladder.
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Fig. 7G —21-year-old woman with inflammatory pseudotumor of the
bladder. Inflammatory pseudotumor manifests as a large multiloculated pelvic
mass such as ovarian tumor. Gross specimen (G) and corresponding
photomicrograph (H) show white, gelatinous mass with cystic change and
hemorrhage. Multifocal infiltrations of various inflammatory cells
(box, H) are evident.
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Fig. 7H —21-year-old woman with inflammatory pseudotumor of the
bladder. Inflammatory pseudotumor manifests as a large multiloculated pelvic
mass such as ovarian tumor. Gross specimen (G) and corresponding
photomicrograph (H) show white, gelatinous mass with cystic change and
hemorrhage. Multifocal infiltrations of various inflammatory cells
(box, H) are evident.
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Fig. 7I —21-year-old woman with inflammatory pseudotumor of the
bladder. Inflammatory pseudotumor manifests as a large multiloculated pelvic
mass such as ovarian tumor. Photomicrographs show spindle cells within
collagenous or myxoid matrix, with a mixed inflammatory cell infiltration. On
smooth muscle actin, tumor cells are diffusely positive (not shown).
(I, H and E x10; J, H and E x40)
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Fig. 7J —21-year-old woman with inflammatory pseudotumor of the
bladder. Inflammatory pseudotumor manifests as a large multiloculated pelvic
mass such as ovarian tumor. Photomicrographs show spindle cells within
collagenous or myxoid matrix, with a mixed inflammatory cell infiltration. On
smooth muscle actin, tumor cells are diffusely positive (not shown).
(I, H and E x10; J, H and E x40)
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Other Rare Locations for Inflammatory Pseudotumor in the Genitourinary Tract
Other rare locations of inflammatory pseudo tumor include the ureter,
prostate, urethra, scrotum, and pelvic or inguinal lymph nodes.
Summary and Conclusion
The imaging characteristics and locations of inflammatory pseudotumor in
the genitourinary tract vary widely, with mostly large masses mimicking
malignant lesions. Inflammatory pseudotumor can also manifest as a multifocal
lesion such as lymphoma (Fig.
2A,
2B) and local tumor recurrence
or metastasis (Fig. 3A,
3B,
3C,
3D). The role of the
radiologist is to ensure that inflammatory pseudotumor is a diagnosis that is
suggested preoperatively and differentiated from malignant lesions by either
particular imaging findings or imaging-guided biopsy. Thus, although definite
radiologic differentiation from malignancy is not clearly possible, we suggest
that familiarity with the manifestations of inflammatory pseudotumor can help
avoid unnecessary radical surgery before histopathologic proof of malignancy
is obtained.
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Abstract
Introduction
