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Rectal Teratoma coexistent with an Ovarian Teratoma: A Case Report

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A teratoma is a mixed tumor containing elements of the three germinal layers. Although teratomas involving the ovary, testicle, and retroperitoneum are frequently found, rectal involvement is extremely rare. There have only been about 50 cases of primary rectal teratoma re- ported since 1864 (1). Herein, we present the radiologic features of a rectal teratoma that was identified with an asymptomatic ovarian teratoma and correctly diagnosed preoperatively using CT images.

Case Report

A 79-year-old woman was referred to our surgical de- partment from a local clinic for the evaluation of a rectal mass. The patient had a history of difficulty in defeca-

tion for the preceding month. The previous medical his- tory of the patient was unremarkable. The results of lab- oratory tests were normal. A hard palpable mass was present upon rectal examination. For the evaluation of the rectal mass, we performed computed tomography (CT) and a barium study.

Post-contrast CT scanning showed an 8×5 cm, well- circumscribed intraluminal rectal mass (Fig. 1A). The rectal mass contained calcifications and fatty compo- nents (Fig. 1B). The rectum was distended by the intra- luminal mass, but no evidence of perirectal fat infiltra- tion or rectal wall thickening was observed. A well-de- marcated cystic ovarian mass with internal calcifica- tions was also seen in the right pelvic cavity on post-con- trast CT scan (Fig. 1B). There was no evidence of rup- ture or infiltration. A double-contrast barium study showed a large, well-defined intraluminal mass located 3-4 cm from the anal verge (Fig. 1C). The distended rectum showed no mucosal destruction or luminal ob- struction.

With the impression of rectal teratoma coexistent with ovarian teratoma, doctors performed surgery on the pa-

J Korean Radiol Soc 2006;54:417-420

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Rectal Teratoma coexistent with an Ovarian Teratoma:

A Case Report

1

Yong Sun Jeon, M.D., Soon Gu Cho, M.D., Sun Keun Choi, M.D.2, In Suh Park, M.D.3, Won Hong Kim, M.D., Kyung Hee Lee, M.D., Chang Hae Suh, M.D.

1Department of Radiology, Inha University College of Medicine

2Department of Surgery, Inha University College of Medicine

3Department of Pathology, Inha University College of Medicine Received November 9, 2005 ; Accepted December 20, 2005 This work was supported by INHA UNIVERSITY Research Grant.

Address reprint requests to : Yong Sun Jeon, M.D., Department of Radiology, Inha University Hospital, 7-206, 3rd St., Shinheung-dong, Choong-gu, Inchon 400-711, Republic of Korea.

Tel. 82-32-890-2767 Fax. 82-32-890-2743 E-mail: [email protected]

We report a case of rectal teratoma coexistent with an ovarian teratoma. To our knowledge, this is the first radiologic report of rectal teratoma. Computed tomography (CT) showed a sharply demarcated cystic and fatty mass with amorphous calcification in the rectum. A double-contrast barium study showed a well-defined intraluminal rectal mass without mucosal destruction. Imaging findings of rectal teratoma allow for correct preoperative diagnosis. CT was helpful in differentiating rectal teratoma from other rectal lesions.

Index words :Rectum, neoplasms Ovary, neoplasms Rectum, CT Teratoma

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tient. In the surgical finding, the rectal mass was soft and ovoid with a stalk protruding into the rectal lumen.

The stalk of the tumor was located in the anterior wall of the rectum, 3 to 4 cm from the anal verge. The tumor

and stalk were transanally resected. The huge pelvic mass in the right ovary was removed by transabdominal right salpingoophorectomy. No relationship was ob- served between the ovarian mass and the rectum at the

Yong Sun Jeon, et al: Rectal Teratoma coexistent with an Ovarian Teratoma

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A B

C

D

E Fig. 1. A. Contrast-enhanced CT scan shows an intraluminal cystic mass (arrow) in the rectum.

B. The upper portion of the rectal mass shows a fat component (white arrow) and small calcifications (arrow). A calcified ovary mass (arrow head) was present in the right pelvic cavity.

C. Double-contrast barium study shows a well-demarcated in- traluminal mass with a smooth surface (arrows). There is no ev- idence of surface destruction or luminal obstruction.

D. Photograph of the resected rectal teratoma specimen shows an ovoid mass with a stalk (arrow) and a skin cover.

E. Photomicrograph of the rectal teratoma shows cartilage (white arrow), adipose tissue (arrow), and lymphoid tissue (thick arrow) (H & E, ×12.5).

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time of surgical excision.

Upon gross examination, the rectal mass was charac- terized as a soft, relatively ovoid mass that was covered with skin and measured 15×9×7 cm (Fig. 1D). The skin was relatively intact. The cut surface showed fi- broadipose tissue and cystic spaces containing a blood clot and tan, friable tissue. Hard bony tissue, measuring 2×0.3 cm, was also present. Upon microscopic exami- nation, the rectal mass was covered with keratinizing squamous epithelium. Abundant adipose tissue, fibrous tissue, irregular blood vessels, nerves, melanin pig- ments, and a small cartilage island were present (Fig.

1E). These findings were compatible with a mature ter- atoma. On gross sectioning, the right pelvic mass was identified as a unilocular cyst containing dark brown, turbid material and measuring 13×11×7 cm.

Microscopic findings showed keratinizing squamous ep- ithelium, hairs, ciliated pseudostratified columnar ep- ithelium, and thyroid tissue. The findings were compati- ble with a benign cystic teratoma of the ovary.

Discussion

It is thought that rectal teratomas may arise from ab- normal germ cells in the embryonic digestive tract (1).

Primordial germ cells are observed at about 3 to 4 weeks of gestation in the endoderm of the umbilical vesicle wall and then move from the dorsal mesentery of the hindgut through the mesenchyme toward the gonadal ridge (1). The adrenal gland, urinary tract, gonads, and rectum are adjoining structures, and the germ cells can potentially enter the rectum aberrantly and create a rec- tal teratoma (1).

Our case revealed a rectal teratoma coexisting with an ovarian teratoma. It is difficult to determine whether the rectal tumor was a primary rectal teratoma or a ter- atoma of an adjacent organ or tissue that had migrated through the rectal wall. Some cases of benign cystic ter- atoma of the ovary that ruptured into the rectum have been reported (2, 3). Peterson et al. (4) collected 1,007 cases of benign cystic teratoma of the ovary, of which rupturing occurred in only 13; this rupturing extends ei- ther into the intraperitoneal cavity or into the adjacent viscus. However, no conclusive decision could be drawn due to the extremely rare pathology in this case.

We postulate that our case is a primary rectal teratoma because of the lack of a relationship between the ovari- an teratoma and the rectum by radiological and surgical findings, the presence of a well-defined stalk, and the

lack of histopathologic evidence of ovarian tissue in the rectal teratoma.

The major clinical symptoms of rectal teratoma are anal bleeding, bloody stools or melena, and constipa- tion. Protrusion of the tumor itself or of hair has been al- so reported occasionally (5). The most common site of primary rectal teratoma growth is the anterior wall of the rectum, as was seen in our case (6). In most reported instances, the primary rectal teratoma itself had pedun- culated character (5, 6).

In our case, the diagnosis could be established preop- eratively by a barium study and cross-sectional CT imaging. The barium study showed an intraluminal mass in the rectum with no evidence of mucosal de- struction or barium leakage. CT scan is particularly well-suited to the diagnostic evaluation of a teratoma be- cause of its high specificity in detecting fat and calcifica- tion. In our case, CT imaging showed the characteristic fat component, small calcifications, and cyst. In a retro- spective study of gastrointestinal teratomas, the borders of the teratomas were round and sharp because of en- capsulation, as was observed in our case (7). The find- ings of imaging and location of the tumor raise the dif- ferential diagnoses that include lipoma, angiomyolipo- ma, lymphangioma, leiomyosarcoma, and abscess.

Lipoma and angiomyolipoma contain fat tissue on CT scans, but are not cystic and do not contain calcifica- tions. Lymphangioma, leiomyosarcoma, and abscess may appear cystic, but the absence of fat or calcification enables the radiologist to differentiate rectal teratoma from these conditions.

In conclusion, we report a rare case of rectal teratoma coexistent with an asymptomatic ovarian teratoma.

Rectal teratoma could be correctly diagnosed by imag- ing features at a preoperative stage. Although very rare, rectal teratoma should be considered in the differential diagnosis of a rectal mass that contains internal fat tissue or calcifications.

References

1. Takao Y, Shimamoto C, Hazama K, Itakura H, Sasaki S, Umegaki E, et al. Primary rectal teratoma: EUS features and review of the literature. Gastrointest Endosc 2000;51:353-355

2. Farkouh E, Allard M, Paquin JG. Benign solid teratoma of the ovary with rupture into the rectum. Can J Surg 1982;25:77-78 3. Landmann DD, Lewis RW. Benign cystic ovarian teratoma with

colorectal involvement. Report of a case and review of the litera- ture. Dis Colon Rectum 1988;31:808-813

4. Peterson WF, Prevost EC, Edmunds FT, Hundley JM Jr, Morris FK. Benign cystic teratomas of the ovary: a clinico-statistical study J Korean Radiol Soc 2006;54:417-420

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of 1,007 cases with a review of the literature. Am J Obstet Gynecol 1955;70:368-382

5. Green JB, Timmcke AE, Mitchell WT Jr. Endoscopic resection of primary rectal teratoma. Am J Surg 1993;59:270-272

6. Tabuchi Y, Tsunemi K, Matsuda T. Variant type of teratoma ap-

pearing as a primary solid dermoid tumor in the rectum: report of a case. Surg Today 1995;25:68-71

7. Bowen B, Ros PR, McCarthy MJ, Olmsted WW, Hjermstad BM.

Gastrointestinal teratomas: CT and US appearance with pathologic correlation. Radiology 1987;162:431-433

Yong Sun Jeon, et al: Rectal Teratoma coexistent with an Ovarian Teratoma

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대한영상의학회지 2006;54:417-420

난소 기형종과 동반된 직장 기형종: 증례 보고1

1인하대학교 의과대학 방사선과학교실

2인하대학교 의과대학 외과학교실

3인하대학교 의과대학 병리학교실

전용선・조순구・최선근2・박인서3・김원홍・이경희・서창해

저자들은 직장 기형종에 관한 첫번째 방사선 증례보고로 난소 기형종과 동반된 직장 기형종 1예를 보고하고자 한다. 바륨 조영 검사에서는 표면의 파괴가 없는 경계가 명확한 직장 내 종양으로 보였으며, CT 소견에서는 직장 내에 경계가 명확한 낭성 종양이 내부에 지방과 석회화를 동반하며 관찰되었다. 영상소견만으로 직장 기형종의 정 확한 수술 전 진단을 내릴 수 있었고, CT가 직장 기형종과 직장의 다른 병변을 감별하는데 도움이 되었다.

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