GYNECOLOGIC
ONCOLOGY
38, 257-261 (1990)
CASE REPORT Squamous Cell Carcinoma Arising in a Cystic Teratoma Presenting as a Vulvar Mass NAOKI TSUKAMOTO, Department
M. D.,’ NOBUHIRO TSURUCHI, M. D., MASASHI IMACHI, M. D., AND TOSHIAKI SAITO, M. D.
of Gynecology and Obstetrics, Kyushu University
Faculty
of Medicine 60, Maidashi 3-I-1, Higashi-ku,
Fukuoka 812, Japan
Received August 29, 1989
up examination. In December 1988, she again felt discomfort and the mass increased in size. When she revisited her gynecologist, the left labial mass was about 10 x 8 x 7 cm. On April 4, 1989, marsupialization of this alleged BarINTRODUCTION tholin’s cyst was attempted. Upon cutting through the Extraovarian teratomas are rarely seen in adults. Their cyst wall, about 200 ml of yellow, greasy fluid mixed occurrence in the female external genitalia is extremely with hair was aspirated. The cyst extended deep up to rare and only a few cases have been reported in children the area of the left ischium. As much cyst wall as pos[ 11. We recently encountered such a case in a 4%year- sible, however incomplete, was removed and the wound old woman. The case is especially interesting in that the was closed. The pathology report on the cyst wall was identified as a squamous cell carcinoma. CT scan pertumor presented as a vulvar mass. formed on the 10th postoperative day revealed a 4 x 5 x 2-cm mass in the left internal obturator muscle (Fig. CASE REPORT I), which was thought to be the unresected cystic mass. The patient was a 4%year-old married Japanesse She was then referred to Kyushu University Hospital woman, gravida 0, para 0. Her menarche was at age 16 for further treatment on May 6, 1989. and menstrual periods were regular. However, she On admission, general physical examination was norunderwent supracervical hysterectomy and left salpingo- mal. Pelvic examination revealed a slightly swollen left oophorectomy for pelvic peritonitis at age 25 and she labium majus. The vaginal mucosa was normal, but an had been amenorrheic since then. approximately 8 x 6 x 3-cm doughy mass was palpated In 1975, when she was 34 years old, she noted several from the left labium to the left pelvic side wall. The small nodules in the surface of her left labium majus. cervix was small and clean. No other pelvic mass was These were removed by a local surgeon, who informed palpated. Tumor markers, including SCC antigen, CEA, her that the nodules were lipomas and that there was AFP, CA-125, and CA-19-9, were all within normal limanother cystic mass located deep in the left labium which its, as were the other blood chemistries. A Pap smear should be observed. In 1983, she noted that the mass of the cervix was normal. had grown to the size of a thumb, but she did not pay On the second day in hospital, the patient developed attention to it because she had no symptoms. signs and symptoms of infection of the left vulvar mass. In October 1988, she noted a gradual increase in the CT scan was performed on May 12, 1989, and showed size of this mass accompanied by pain and consulted a a cystic mass extending from the left internal obturator local gynecologist. It was diagnosed as infected Bar- muscle to the left labial region (Fig. 2). The cystic mass tholin’s cyst and was treated with antibiotics and anti- close to the labium was interpreted as an abscess, which inflammatory drugs. She did not go back for a follow- ruptured spontaneously on the day of the CT scan. Figure 3 is a photograph of the vulva after rupture of the ’ To whom reprint requests should be addressed. infected cystic mass. A case of squamous cell carcinoma arising in a cystic teratoma presenting as a vulvar mass is reported. This appears to be the first repOrt of such a case. o 1990 Academic press, I~C.
257 OO!W8258/90 $I .50 Copyright 0 1990by AcademicPress,Inc. All rights of reproductionin any form reserved.
258
TSUKAMOTO
ET AL
FIG. 1. The 4 x 5 x 2-cm mass observed in the left internal obturator muscle.
FIG. 2. The mass extended from the left internal obturator muscle to the left labial region (arrow). Fine linear shadows around the mass were suggestive of inflammatory reaction and/or tumor infiltration.
259
CASE REPORT
FIG. 3. Swollen left vulva. The white spot in the center indicates the opening of the ruptured abscess.
On May 24, 1989, the patient underwent surgery. At first, the abdomen was opened and paraaortic lymph node biopsy, bilateral pelvic lymph node dissection, right salpingo-oophorectomy, trachelectomy were performed. Bilateral groin lymph node dissection, radical vulvectomy, and resection of the left pelvic side wall mass followed. The mass was easily separated from the walls of the vagina and rectum. However, it was firmly attached to the inner aspect of the left ischial bone and had to be separated, using an electro-cautery, from above and from below. Figure 4 shows the extirpated vulva and tumor.
Pathologic examination revealed no lymph node metastases. The cervix and right ovary were also normal. The resected and collapsed cystic tumor measured 7 x 3 x 3 cm and its boundary from the surrounding fatty tissue was unclear. The inner surface epithelium of the cyst wall was mostly sloughed and infiltrated with chronic inflammatory cells and abundant foreign body giant cells (Fig. 5). The cyst wall was partly covered by benign keratinizing squamous epithelium with skin appendages, such as sebaceous glands, sweat glands, and hair shafts (Fig. 6). The cyst wall was also covered by invasive squamous cell carcinoma deeply infiltrating the stroma (Fig. 6 and 7). Her postoperative course was uneventful. She received 40 Gy of external irradiation to the left pelvic side wall from June 14 to July II, 1989, and was discharged in satisfactory condition on July 13, 1989.
DISCUSSION Teratomas are said to develop from germ cells. Germ cells originate in the yolk sac and migrate to the gonadal sites by amoeboid movements. Teratomas can develop anywhere along the migration route of germ cells and their extragonadal occurrences are reported in the anterior mediastinum, retroperitoneal region, presacral and coccygeal areas, intracranial sites, neck and abdominal viscera [2]. However, there have been no detailed accounts of a vulvar teratoma [3]. To our knowledge, our
FIG. 4. Extirpated vulva and tumor (arrow). The lower end of the tumor was attached to the left ischial bone
260
TSUKAMOTO
ET AL.
FIG. 5. The surface epithelium of the cyst wall was sloughed and the stroma was infiltrated with chronic inflammatory cells and multiple body giant cells. H & E, x38.
FIG. 6. Part of the cyst wall was covered by benign keratinized squamous epithelium. Sebaceous and sweat glands are seen in the left lower field and invasive squamous carcinoma in the right upper field (arrows). H & E, ~21.
261
CASE REPORT
FIG. 7. High-power magnification of a focus of invasive squamous cell carcinoma. H & E.
case seems to be the first of such a case presenting as a vulvar mass. The patient noted the presence of the left vulvar mass since age 34. During the following 14 years, the mass gradually increased in size. When she finally sought medical attention because of pain and swelling, it was a squamous cell carcinoma. Judging from her history and the pathologic findings (Figs. 5 and 6), it can be speculated that this patient originally had a benign mature cystic teratoma which showed malignant transformation in due course. Concerning the original site of this tumor, it is possible that this tumor developed retroperitoneally at first arising in the region of the obturator space or obturator muscle (Figs. 1 and 2) and grew up extending
x
106.
to the left labial region. However, it is not known if the patient had a left side wall mass before or not, because she had no pelvic examination since her surgery at age 25. REFERENCES 1, Huffman, J. W., Dewhurst, C. J., and Capraro, V. J. The gynecology und adolescence, Saunders, Philadelphia, 2nd ed., p. 231 (1981). 2. Engel. R. M., Elkins, R. C. and Fletcher, B. D. Retroperitoneal teratoma: Review of the literature and presentation of an unusual case, Cancer 22, 1068-1073 (1968). 3, Fox, H., and Buckley, C. H. Non-epithelial and mixed turnouts of the vulva, in The vu/vu (C. M. Ridley. Ed.). Churchill Livingstone, Edinburgh, p. 256 (1988). of‘ childhood
ONCOLOGY
38, 257-261 (1990)
CASE REPORT Squamous Cell Carcinoma Arising in a Cystic Teratoma Presenting as a Vulvar Mass NAOKI TSUKAMOTO, Department
M. D.,’ NOBUHIRO TSURUCHI, M. D., MASASHI IMACHI, M. D., AND TOSHIAKI SAITO, M. D.
of Gynecology and Obstetrics, Kyushu University
Faculty
of Medicine 60, Maidashi 3-I-1, Higashi-ku,
Fukuoka 812, Japan
Received August 29, 1989
up examination. In December 1988, she again felt discomfort and the mass increased in size. When she revisited her gynecologist, the left labial mass was about 10 x 8 x 7 cm. On April 4, 1989, marsupialization of this alleged BarINTRODUCTION tholin’s cyst was attempted. Upon cutting through the Extraovarian teratomas are rarely seen in adults. Their cyst wall, about 200 ml of yellow, greasy fluid mixed occurrence in the female external genitalia is extremely with hair was aspirated. The cyst extended deep up to rare and only a few cases have been reported in children the area of the left ischium. As much cyst wall as pos[ 11. We recently encountered such a case in a 4%year- sible, however incomplete, was removed and the wound old woman. The case is especially interesting in that the was closed. The pathology report on the cyst wall was identified as a squamous cell carcinoma. CT scan pertumor presented as a vulvar mass. formed on the 10th postoperative day revealed a 4 x 5 x 2-cm mass in the left internal obturator muscle (Fig. CASE REPORT I), which was thought to be the unresected cystic mass. The patient was a 4%year-old married Japanesse She was then referred to Kyushu University Hospital woman, gravida 0, para 0. Her menarche was at age 16 for further treatment on May 6, 1989. and menstrual periods were regular. However, she On admission, general physical examination was norunderwent supracervical hysterectomy and left salpingo- mal. Pelvic examination revealed a slightly swollen left oophorectomy for pelvic peritonitis at age 25 and she labium majus. The vaginal mucosa was normal, but an had been amenorrheic since then. approximately 8 x 6 x 3-cm doughy mass was palpated In 1975, when she was 34 years old, she noted several from the left labium to the left pelvic side wall. The small nodules in the surface of her left labium majus. cervix was small and clean. No other pelvic mass was These were removed by a local surgeon, who informed palpated. Tumor markers, including SCC antigen, CEA, her that the nodules were lipomas and that there was AFP, CA-125, and CA-19-9, were all within normal limanother cystic mass located deep in the left labium which its, as were the other blood chemistries. A Pap smear should be observed. In 1983, she noted that the mass of the cervix was normal. had grown to the size of a thumb, but she did not pay On the second day in hospital, the patient developed attention to it because she had no symptoms. signs and symptoms of infection of the left vulvar mass. In October 1988, she noted a gradual increase in the CT scan was performed on May 12, 1989, and showed size of this mass accompanied by pain and consulted a a cystic mass extending from the left internal obturator local gynecologist. It was diagnosed as infected Bar- muscle to the left labial region (Fig. 2). The cystic mass tholin’s cyst and was treated with antibiotics and anti- close to the labium was interpreted as an abscess, which inflammatory drugs. She did not go back for a follow- ruptured spontaneously on the day of the CT scan. Figure 3 is a photograph of the vulva after rupture of the ’ To whom reprint requests should be addressed. infected cystic mass. A case of squamous cell carcinoma arising in a cystic teratoma presenting as a vulvar mass is reported. This appears to be the first repOrt of such a case. o 1990 Academic press, I~C.
257 OO!W8258/90 $I .50 Copyright 0 1990by AcademicPress,Inc. All rights of reproductionin any form reserved.
258
TSUKAMOTO
ET AL
FIG. 1. The 4 x 5 x 2-cm mass observed in the left internal obturator muscle.
FIG. 2. The mass extended from the left internal obturator muscle to the left labial region (arrow). Fine linear shadows around the mass were suggestive of inflammatory reaction and/or tumor infiltration.
259
CASE REPORT
FIG. 3. Swollen left vulva. The white spot in the center indicates the opening of the ruptured abscess.
On May 24, 1989, the patient underwent surgery. At first, the abdomen was opened and paraaortic lymph node biopsy, bilateral pelvic lymph node dissection, right salpingo-oophorectomy, trachelectomy were performed. Bilateral groin lymph node dissection, radical vulvectomy, and resection of the left pelvic side wall mass followed. The mass was easily separated from the walls of the vagina and rectum. However, it was firmly attached to the inner aspect of the left ischial bone and had to be separated, using an electro-cautery, from above and from below. Figure 4 shows the extirpated vulva and tumor.
Pathologic examination revealed no lymph node metastases. The cervix and right ovary were also normal. The resected and collapsed cystic tumor measured 7 x 3 x 3 cm and its boundary from the surrounding fatty tissue was unclear. The inner surface epithelium of the cyst wall was mostly sloughed and infiltrated with chronic inflammatory cells and abundant foreign body giant cells (Fig. 5). The cyst wall was partly covered by benign keratinizing squamous epithelium with skin appendages, such as sebaceous glands, sweat glands, and hair shafts (Fig. 6). The cyst wall was also covered by invasive squamous cell carcinoma deeply infiltrating the stroma (Fig. 6 and 7). Her postoperative course was uneventful. She received 40 Gy of external irradiation to the left pelvic side wall from June 14 to July II, 1989, and was discharged in satisfactory condition on July 13, 1989.
DISCUSSION Teratomas are said to develop from germ cells. Germ cells originate in the yolk sac and migrate to the gonadal sites by amoeboid movements. Teratomas can develop anywhere along the migration route of germ cells and their extragonadal occurrences are reported in the anterior mediastinum, retroperitoneal region, presacral and coccygeal areas, intracranial sites, neck and abdominal viscera [2]. However, there have been no detailed accounts of a vulvar teratoma [3]. To our knowledge, our
FIG. 4. Extirpated vulva and tumor (arrow). The lower end of the tumor was attached to the left ischial bone
260
TSUKAMOTO
ET AL.
FIG. 5. The surface epithelium of the cyst wall was sloughed and the stroma was infiltrated with chronic inflammatory cells and multiple body giant cells. H & E, x38.
FIG. 6. Part of the cyst wall was covered by benign keratinized squamous epithelium. Sebaceous and sweat glands are seen in the left lower field and invasive squamous carcinoma in the right upper field (arrows). H & E, ~21.
261
CASE REPORT
FIG. 7. High-power magnification of a focus of invasive squamous cell carcinoma. H & E.
case seems to be the first of such a case presenting as a vulvar mass. The patient noted the presence of the left vulvar mass since age 34. During the following 14 years, the mass gradually increased in size. When she finally sought medical attention because of pain and swelling, it was a squamous cell carcinoma. Judging from her history and the pathologic findings (Figs. 5 and 6), it can be speculated that this patient originally had a benign mature cystic teratoma which showed malignant transformation in due course. Concerning the original site of this tumor, it is possible that this tumor developed retroperitoneally at first arising in the region of the obturator space or obturator muscle (Figs. 1 and 2) and grew up extending
x
106.
to the left labial region. However, it is not known if the patient had a left side wall mass before or not, because she had no pelvic examination since her surgery at age 25. REFERENCES 1, Huffman, J. W., Dewhurst, C. J., and Capraro, V. J. The gynecology und adolescence, Saunders, Philadelphia, 2nd ed., p. 231 (1981). 2. Engel. R. M., Elkins, R. C. and Fletcher, B. D. Retroperitoneal teratoma: Review of the literature and presentation of an unusual case, Cancer 22, 1068-1073 (1968). 3, Fox, H., and Buckley, C. H. Non-epithelial and mixed turnouts of the vulva, in The vu/vu (C. M. Ridley. Ed.). Churchill Livingstone, Edinburgh, p. 256 (1988). of‘ childhood
