A Case of Incidentally Found Bilateral Retroperitoneal Cavernous Hemangiomas

doi:10.4236/oju.2011.13012

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Open Journal of Urology, 2011, 1, 56-59

doi:10.4236/oju.2011.13012 Published Online August 2011 (http://www.SciRP.org/journal/oju)

A Case of Incidentally Found Bilateral Retroperitoneal

Cavernous Hemangiomas

Shunsuke Yui1, Masayoshi Zaitsu1, Koji Mikami1, Y ut a Take shi ma1, Akiko Tonooka2,

Toshimasa Uekusa2, Takumi Takeuchi1

1Department of Ur ol o gy, Kanto Rosai Hospital, Kawasaki, Japan

2Pathology, Kanto Rosai Hospital, Kawasaki, Japan

E-mail: [email protected]

Received July 5, 2011; revised July 31, 2011; accepted August 9, 2011

Abstract

A 59-year-old male was admitted to our hospital because of incidentally found right retroperitoneal tumor.

He had undergone removal of a hemangioma in the left oral cavity four years before. An abdominal CT scan

performed in our hospital revealed poorly enhanced bilateral retroperitoneal tumors adjacent to kidneys.

Those tumors were of low signal intensity on T1-weighted images and high on T2-weighted images by

magnetic resonance imaging. The right retroperitoneal tumor of 2.5 cm in size was surgically removed and

histopathological examination indicated cavernous hemangioma. The smaller left retroperitoneal tumor of

1.1 cm in size was left untouched to be followed up, as we supposed that it has the same benign pathology.

There have been no previous cases of retroperitoneal cavernous hemangioma as a presentation of multiple

hemangiomas.

Keywords: Hemangioma, Bilateral, Retroperitoneum

1. Introduction

Primary retroperitoneal tumors just account for 0.2% of

all tumors in a old article publish ed in 1954 [1] and there

are no available reports definitely describing the recent

prevalence of retroperitoneal tumors. Braasch et al. col-

lected 101 cases of retroperitoneal tumor from the litera-

ture and reported in 1967 that more than 80% of cases

were malignant and symptomatic [2]. Nowadays benign,

smaller, and less symptomatic retroperitoneal tumors

must be diagnosed with the improvement and prevalence

of advanced imaging modalities as CT scan, magnetic

resonance imaging, and ultrasonography. Nakajima et al.

reported in 1997 that 10 (40%) out of 25 retroperitoneal

tumors were malignant [3], thus the percentage of ma-

lignancy among all diagnosed retroperitoneal tumors

seems decreased than before.

Retroperitoneal hemangioma is very uncommon,

while the true incidence of that disorder among retrop-

eritoneal tumors is basically unknown, as most of them

may be asymptomatic and remain undiagnosed. That

there have been only few reports of retroperitoneal he-

mangioma diagnosed only with radiological imagings [4]

may indicate that surgical procedure has been chosen for

both diagnostic and therapeutic purposes. Here we show

a case of bilateral retroperitoneal cavernous hemangio-

mas which was difficult to be diagnosed before surgical

extirpation.

2. Case Report

A 59-year-old male was admitted to our hospital because

of incidentally found right retroperitoneal tumor. Tumor

markers as well as regular laboratory data were unre-

markable. Urine cytology was negative. An abdominal

CT scan performed in our hospital revealed a right re-

troperitoneal tumor of 2.5 cm in size and a left retroperi-

toneal tumor of 1.1 cm in size adjacent to either kidney.

Those tumors with speckled calcification were not en-

hanced so much both in early phase and late phase (Fig-

ure 1). By an abdominal magnetic resonance imaging,

those tumors were of low signal intensity on T1-

weighted images and high on T2-weighted images (Fig-

ure 2). We have decided to extirpate the right retroperi-

toneal tumor using transperitoneal approach, because

those tumors could not be determined to be of benign

pathology by imaging studies. We did not adopt lararo-

scopic and retroperitoneoscopic surgery because of un-

S. YUI ET AL.

known pathology.

At operation, there was little adhesion between the

tumor and the surrounding structures. The tumor was

supposed to be originated from the vascular sheath of the

right renal artery and there were small feeders from the

sheath. The tumor was irregular on surface and reddish

like a raspberry in color (Figure 3). We utilized renal

arterial clump for a short time in order to reduce gradual

bleeding from the tumor surface which occurred when

the preparation of the tumor was initiated. The resected

tumor was 3.0 × 1.5 × 1.5 cm in size. The cut section

showed sponge-like appearance containing solid com-

ponent (Figure 4).

Histology of the tumor (Figure 5) was diagnosed as

cavernous hemangioma composed of elaborately intera-

nastomosing vascular spaces of large dilated blood-filled

vessels. Vascular channels with fibrous walls were lined

by flattened endothelial cells which have no atypism.

The smaller left retroperitoneal tumor of 1.1 cm in size

was left untouched to be followed up, as we supposed

that it has the same benign pathology.

3. Discussion

We have experienced a relatively rare case of bilateral

retroperitoneal cavernous hemangiomas occurred in an

elder man. Hemangioma is often found in infants, espe-

cially in the skin [5]. Hemangioma is histologically de-

fined as a benign tumor or malformation made up of

mature well-formed vessels usually lined by a single

layer of endothelium and is classified into six categories,

i.e. capillary hemangioma, cavernous hemangioma, ve-

nous hemangioma, epithelioid hemangioma, pyogenic

Figure 1. Example of a figure caption A CT scan demon-

strating bilateral retroperitoneal tumors. Arrows indicate

tumors.

Figure 2. An MR imaging demonstrating bilateral retro-

peritoneal tumors. Arrows indic a te tumors.

Figure 3. A photograph during operation. T: right retro-

peritoneal tumor, A: taped right renal artery, V: right renal

vein, K: right kidney.

Figure 4. A macroscopic cut section of the right retro-

pritoneal tumor.

granuloma, and acquired tirfted hemangioma [6]. He-

mangioma is also known to be found in the oral region

[7] as was observed in the present case.

In the present case, hemangiomas were multiple in bi-

lateral retroperitoneal spaces as well as in the mouth.

Multiple hemangiomas are often a part of established

syndromes such as Sturge-Weber syndrome, Maffucci

syndrome, Kasabach-Merritt syndrome, and von Hippel-

58 S. YUI ET AL.

Figure 5. Hematoxylin and eosin staining of the right re-

troperitoneal tumor showing cavernous hemangioma.

Lindau disease, but this case did not seem to apply to

them as it lacks in the characteristics of those disorders.

There have been no previous cases of retroperitoneal

cavernous hemangioma as a presentation of multiple

hemangiomas.

Surgical removal of retroperitoneal tumors is indicated

as long as the possibility of poten tial malignan cy remains.

Maximum diameter of malignant retroperitoneal tumors

is reported to be larger than that of benign ones (11.45 ±

1.90 cm vs 5.31 ± 0.43 cm) [3]. The tumor size of the

present case was relatively small and the probability of

malignancy was not very high in the view points of tu-

mor size, then we might have followed up the interval

change in the tumor size before indicating surgery. Addi-

tionally, retroperitoneal hemangiomas have a risk of

rupture and bleeding [8] even they are histologically be-

nign, leading to a rational of choosing surgical extirpa-

tion especially when they are large. Although we adopted

open surgical approach, laparoscopic resection of retrop-

eritoneal cavernous hemangioma has been reported

[9,10]. It may be appropriate to remove retroperitoneal

hemangiomas laparoscopically or retroperitoneoscopi-

cally if they are probably benign .

Besides surgical treatments, there are some options as

corticosteroid therapy and radiotherapy in the treatment

of hemangioma. While corticosteroid therapy is mainly a

choice of treatments of capillary hemangioma [11], there

is a report of unresectable 8 × 5 cm retroperitoneal cav-

ernous hemangioma diagnosed in a female neonate 20

days after birth and showing anemia and thrombocyto-

penia, which disappeared following oral corticosteroid

therapy [12]. Radiotherapy is also given to treat symp-

tomatic hemangiomas [13].

In the present case, the smaller left retroperitoneal tu-

mor of 1.1 cm in size was left untouched to be followed

up, as we supposed that it has the same ben ign pathology.

When the left retroperiton eal tumor grows larger enough ,

we may need to re-consider if he needs some treatment

including surgery.

4. References

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[11] M. T. Edgerton, “The Treatment of Hemangiomas: With

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doi:10.1097/00000658-197605000-00009

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doi:10.1016/0022-3468(73)90296-0

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