Case Report
Inflammatory Myofibroblastic Tumor of
Spermatic Cord in Undescended Testis
Chi-Hang Yee, Ka-Fai To, See-Ming Hou, and Chi-Fai Ng
Inflammatory myofibroblastic tumor (IMT) is an uncommon condition and only a few cases of IMT in the spermatic
cord have been reported, with none associated with an undescended testis. We present the case of a man with an
undescended left testis who had an IMT involving the spermatic cord. He was treated with surgery and no adjuvant
therapy afterward. He was disease free at 2.5 years of follow-up. A brief discussion of the pathologic features and
management of the condition is also provided. UROLOGY 73: 1423.e9 –1423.e12, 2009. © 2009 Elsevier Inc.
I
nflammatory myofibroblastic tumor (IMT) is an un-
common condition and was initially described in the
lungs.
1
It is composed of myofibroblastic mesenchy-
mal spindle cells with an inflammatory infiltrate of
plasma cells, lymphocytes, and eosinophils.
2
Cases have
been reported in other sites, including the retroperito-
neum,
3
gastrointestinal tract,
4
and genitourinary tract.
However, only a few case of IMT in the spermatic cord
have been reported,
5–7
with none associated with an
undescended testis. We present the case of a man with an
undescended testis who had an IMT involving the sper-
matic cord.
CASE REPORT
A 40-year-old man, with a known undescended left testis,
presented with a left lower quadrant mass that he had
noted a few weeks earlier. All laboratory values were
unremarkable, including -fetoprotein, -human chori-
onic gonadotropin, and lactate dehydrogenase. Com-
puted tomography demonstrated an 8 7 10 cm
heterogeneous contrast-enhancing mass in the left iliac
crest, with a stalk going through the left inguinal canal;
no left testis was identified. Positron emission tomogra-
phy revealed that the mass was hypermetabolic, with a
standardized glucose uptake value of 5.2, compatible with
a malignant tumor (Fig. 1). No evidence of lymph node
or distant metastasis was found.
Laparotomy was performed, and a well-encapsulated
intra-abdominal mass arising from the left spermatic cord
was found. The left undescended testis was also located
intra-abdominally and appeared normal. The tumor mass
was excised en bloc with the testis. On macroscopic
examination, the mass appeared to be a solid, firm, white
tumor, separate from the testis. No gross necrosis or
hemorrhage was noted (Fig. 2). Examination of the mi-
croscopic section showed a spindle cell proliferative le-
sion arranged mainly in a short fascicular pattern among
a fibrous stroma, rich in mixed inflammatory cells. The
cells exhibited mildly pleomorphic nuclei, with occa-
sional large lobulated nuclei and distinct nucleoli. The
mitotic activity was low (Fig. 3). The resection margins
were clear. Immunohistochemically, the tumor was pos-
itive for smooth muscle actin. Staining for desmin, ana-
plastic lymphoma kinase (ALK) 1, CD34, CD21, cyto-
keratins (AE1/AE3), and S100 were negative. In situ
hybridization for Epstein-Barr virus was negative. The
findings were consistent with an IMT arising from the
spermatic cord.
No adjuvant treatment was given after surgical exci-
sion, and he was disease free at 2.5 years of follow-up.
COMMENT
IMT has been described in reported studies with a variety
of names, including inflammatory pseudotumor, atypical
myofibroblastic tumor, pseudosarcoma, and proliferative
funiculitis.
8
The term “proliferative funiculitis” was first
proposed in 1992 and denoted an inflammatory myofi-
broblastic tumor involving the spermatic cord.
5
It is a
rare, low-grade malignant tumor with myofibroblastic
differentiation.
9
Only a few cases have been reported
6,7
after the initial reports of 5 cases of this entity in the
spermatic cord.
5
Extrapulmonary IMT is generally ob-
served to be a disease of young people.
1
However, from
the published data, proliferative funiculitis preferentially
affects men of middle to advanced age,
5–7
in line with our
case. On computed tomography, IMT displays no specific
density values. Variable enhancement occurs after ad-
ministration of iodinated contrast material.
10
On mag-
netic resonance imaging, IMT demonstrates low-signal
intensity on T
1
-weighted images and frequently on T
2
-
weighted images. Usually marked enhancement is seen
after gadolinium administration.
11
From the Department of Surgery and Department of Anatomical and Cellular Pathol-
ogy, Chinese University of Hong Kong, Hong Kong, People’s Republic of China
Address for correspondence: Chi-Fai Ng, M.D., Department of Surgery, Chinese
University of Hong Kong, Prince of Wales Hospital, Hong Kong, People’s Republic of
China. E-mail: ngcf@surgery.cuhk.edu.hk
Submitted: January 25, 2008, accepted (with revisions): April 9, 2008
© 2009 Elsevier Inc. 0090-4295/09/$34.00 1423.e9
All Rights Reserved doi:10.1016/j.urology.2008.04.019