Journal of Obstetrics and Gynaecology
ISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20
Vaginal teratoma: A case report and review of the literature F. Vural, B. Vural & N. Paksoy To cite this article: F. Vural, B. Vural & N. Paksoy (2015) Vaginal teratoma: A case report and review of the literature, Journal of Obstetrics and Gynaecology, 35:7, 757-758, DOI: 10.3109/01443615.2015.1004525 To link to this article: http://dx.doi.org/10.3109/01443615.2015.1004525
Published online: 11 Feb 2015.
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Date: 19 October 2015, At: 08:11
Gynaecology Case Reports 757
Vaginal teratoma: A case report and review of the literature F. Vural1, B. Vural2 & N. Paksoy3 1Department of Obstetrics and Gynecology, Haydarpaşa Numune
Teaching Hospital, Istanbul, Turkey, 2Department of Obstetrics and Gynecology, Kocaeli University Faculty of Medicine, Kocaeli, Turkey, and 3Department of Pathology, Kocaeli University Faculty of Medicine, Kocaeli, Turkey DOI: 10.3109/01443615.2015.1004525 Correspondence: Fisun Vural, M.D., Orhan mah. Bağtepe Sok. Erenkaya park Evleri A2/2 Bağçeşme/Kocaeli, Turkey. E-mail: [email protected]
Downloaded by [University of Otago] at 08:11 19 October 2015
Introduction
Dermoid cysts (benign cystic teratoma) are benign germ-cell tumours that contain well-differentiated derivatives of all three germ layers (Sharma et al. 2012). They usually occur along the line of embryonic fusion over the head and neck region, the mediastinum and the presacral area and are present at birth or develop in early childhood. Although most teratomas occur in the midline, they may present atypically as vaginal dermoid cysts (Sharma et al. 2012; Siu et al, 2003). A true cutaneous dermoid cyst is formed by ectodermal cells buried along the lines of closure of embryonic clefts and sinuses by skin fusion during the embryonic stage, these cells are able to transform into hair, skin and appendages. If dermoid cyst contains mesodermal cells, bone, teeth and cartilage may develop. If teeth are found in the vaginal dermoid cyst, it is known as ‘vagina dentata’ (Sharma et al. 2012; Siu et al, 2003). Mention of vagina dentata is found in the creation myths and folk stories of many cultures such as Native Americans in North America and different populations in Russia, Japan, India, Samoa and New Zealand. After the 19th century, these stories were interpreted as the ‘castration anxiety of man’ (Jackson, 1971). Although vagina dentata is a universal symbol of men’s fear of women, vaginal dermoid cyst is quite rare. To the best of our knowledge, there are 7 cases of vaginal dermoid cysts published thus far in the English literature (Sharma et al. 2012). We present a case of a vaginal dermoid cyst along with a review of the literature.
Case report Clinical history A 15-year-old girl presented with a protruding mass from the hymenal ring. Her height was 160 cm and she weighed 40 kg. She
Figure 1. Microscopic appearance of vaginal teratoma. The dilated mature glandular tissue and mature cartilage tissue. (Figure 1. haematoxylin and eosin; 125).
had a history of regular menstrual cycles, was Tanner stage 5 in breast development and had a normal body hair growth pattern. There was no history of in utero exposure to diethylstilbestrol, radiotherapy or any medical disorders. The ultrasonographic findings of the pelvis were normal: uterus (60 55 45 mm), right ovary (25 20 20 mm) and left ovary (20 18 15 mm) with normal follicular appearance. Gynaecological examination revealed both soft and semisolid, polypoid mobile structures with a smooth surface protruding through the introitus (hymenal ring). The polypoid tissue measured 5 4 3 cm with a stalk that was 0.5 cm in width attached to the lower lateral wall of the vaginal submucosal tissue. Complete excision of the mass was performed under local anaesthesia. The patient’s post-operative course was uneventful.
Histopathological findings Macroscopically, a polypoid structure measuring 5 4 3 cm was described with necrotic margins. The cut surface demonstrated three well-demarcated areas of cartilage tissue embedded within the structure. The histopathological examination revealed a polypoid tissue covered by squamous epithelium with some reactive changes. Subepithelial connective tissue contained irregular-shaped, thin-walled, dilated glandular structures and a few islands of mature cartilage tissue. There were no other adult tissues. No immature elements were also seen. The diagnosis was a mature teratoma, shown in Figure 1.
Discussion
Vaginal cysts are not uncommon findings in clinical practice – epidermal inclusion cysts, Gardner’s duct cysts and Bartholin’s duct cysts are common types. However, dermoid cyst, ectopic ureterocele and paraurethral cyst is rare types (Siu et al, 2003). Dermoid cysts are a special form of mature teratoma and they represent the most common benign germ-cell tumour and the most common neoplasms of the ovary in young women (Sharma et al. 2012). Dermoid cysts are usually present at birth and continue to grow in size with age. The cyst is similar to those present on skin tissue and can contain fat, and occasionally hair, bone, nails, teeth, eyes, cartilage and thyroid tissue. On occasion, teeth can be found within the vaginal dermoid cyst, which is why they are called ‘vagina dentata’ (Sharma et al. 2012; Siu et al, 2003). Although vagina dentata appears in the creation of myths and folkloric stories of many cultures (Jackson, 1971), a dermoid cyst of the vagina is quite rare. Vaginal dermoid cysts were first described in 1899 by Stokes, who reported a 44-year-old woman who underwent removal of a 1-cm cyst located within the hymen. The cyst contained numerous sebaceous glands and a few hair follicles (Stokes 1899). Curtis (1913) described an ulcerated orange-sized necrotic cyst containing hair and sebaceous materials in the vaginal mucosa. In another case report, a 4-inch cyst was expelled from a woman’s vagina following delivery of her second child. The cyst contained thick sebaceous material with matted hair and had been attached to the vaginal wall by a narrow stalk (Johnston 1939). In still another report, excision of a painful right vaginal wall cyst revealed it to be a dermoid cyst confirmed by histopathological examination (Hirose et al, 1991). Subsequently, Siu et al (2003), AlShahrani and Heywood (2006) and Sharma et al. (2012) reported three cases of paravaginal dermoid cysts. Our case differed from the others in that 1) the girl was an adolescent; 2) the mass was a smooth-based multiple polypoid mass with a stalk and 3) the cyst was not located in the paravaginal space but rather protruded from the hymen. Siu et al (2003) noted that cutaneous dermoid cysts and epidermal inclusion cysts of the vagina had similar characteristics. During the repair of an episiotomy, skin appendages may become buried in the wound leading to features that are consistent with a dermoid cyst. However, this latter case involved a 15-year-old adolescent girl with no history of sexual intercourse. Thus, the aetiology of vaginal dermoids remains to be elucidated. Vaginal teratoma is a rare and benign condition that can be diagnosed by histopathological examination and treated with surgical excision. Declaration of interests: The authors report no declarations of interest. The authors alone are responsible for the content and writing of the paper.
758 Gynaecology Case Reports
References
Downloaded by [University of Otago] at 08:11 19 October 2015
Al-Shahrani M, Heywood M. 2006. A rare case of vaginal dermoid cyst: a case report and review of literature. Biomedical Research 17:95–97. Curtis AH. 1913. Transactions of societies. Chicago Gynecological Society. Surgery, Gynecology and Obstetrics 16:715. Hirose R, Imai A, Kondo H, Itoh K, Tamaya T. 1991. A dermoid cyst of the paravaginal space. Archives of Gynecology and Obstetrics. 249:39–41. Jackson B. 1971. Vagina dentata and cystic teratoma. Journal of American Folklore 84:341–342. Johnston HW. 1939. A dermoid cyst of the vagina complicated by pregnancy. Canadian Medical Association Journal 41:386. Sharma R, Rao KA, Nagraj S. 2012. A rare case of vaginal dermoid cyst: a case report and review of literature. International Journal of Infertility and Fetal Medicine 3:102–104. Siu SS, Tam WH, To KF, Yuen PM. 2003. Is vaginal dermoid cyst a rare occurrence or a misnomer? A case report and review of the literature. Ultrasound in Obstetrics & Gynecology 21:404–406. Stokes JE. 1899. The etiology and structure of true vaginal cysts. Johns Hopkins Hospital Reports 7:109–136.
A rare case of primary clear-cell adenocarcinoma of the bladder arising from bladder endometriosis V. Loizzi1,2, G. Cormio1,2, L. Leone1, M. Falagario1, S. Longo3, L. Resta4 & L. Selvaggi1 1Department of Obstetrics, Gynecology and Neonatology, University
of Bari, Bari, Italy, 2Division of Gynecology Oncology, IRCCS Bari, University of Bari, Bari, Italy, 3Department of Internal Medicine, University of Bari, Bari, Italy, and 4Department of Pathology, University of Bari, Bari, Italy DOI: 10.3109/01443615.2015.1004526 Correspondence: Vera Loizzi, M.D., Aggregate Professor, Department of Obstetrics, Gynecology and Neonatology, University of Bari, Piazza Giulio Cesare 11, 70124 Bari, Italy. E-mail: [email protected]
Introduction
Endometriosis is a common benign gynaecological condition affecting 5–15% of women in their reproductive age and only 3–5% of post-menopausal women, in which endometrial tissue grows outside uterine cavity, leading to pelvic pain and infertility (Vignali et al. 2002). Even if endometriosis is a benign condition, it can spread to other organs by invading and disrupting their tissues, sharing these characteristics with invasive cancers. It is well known that ovarian cancer, especially endometrioid and clear cell types, can be the result of a malignant transformation of foci of endometriosis (Bacci et al. 2009), but even if extra-gonadic endometriosis malignant transformation was considered an exceptional event, recent studies suggested that it can also be associated with other kinds of cancer (Ness 2003; Olson et al. 2002; Scully et al. 1966). Primary clear-cell bladder adenocarcinoma is a very rare tumour, affecting mostly women; it can be associated with bladder endometriosis suggesting a mullerian origin of this rare cancer (Lu et al. 2012).
of the bladder. The patient underwent a surgery with a laparotomic approach. An extended pelvic disease was observed. Only uterine fundus was visible because bladder was completely adherent to its anterior wall, and posteriorly adnexa were fixed to posterior uterine wall. An approximately 8-cm mass involving the whole bladder vault was evaluated. A partial cystectomy with removal of left ovarian cyst was performed. The final pathology report revealed a surrounding endometriosis zone in which we observed a tumour involving the bladder wall with mucosal saving (Figure 1). This tumour is composed of an aggregate of glandular structures with lining monofilament epithelial that in some areas appear flattened or cubical, whereas in others it is composed of micropapillary protrusions showing an axis more often hyaline and prominent vesicular nuclei on the epithelium and a large nucleolus. There were sporadic calcified concretions such as psammoma bodies. The periodic acid–Schiff reaction with diastase showed occasional presence of intracytoplasmic glycogen. According to Sampson’s criteria, diagnosis of clear-cell adenocarcinoma (CCA) of the bladder arisen from bladder endometriosis was made. The absence of ovarian and endometrial involvement leads us to believe that neoplasm should be a primary tumour in the bladder wall. The patient underwent 4 cycles of chemotherapy with paclitaxel, cisplatin and epirubicin (TEP). Afterwards a laparotomy with bilateral pelvic lymphadenectomy, radical Piver type II hysterectomy and bilateral salpingo-oophorectomy was performed. The pathology report revealed the presence of CCA in the pelvic lymph nodes, uterine serosa and ovaries. Other two cycles of TEP and external pelvic radiotherapy, with a total dose of 4500 cGy, were then given to the patient. The patient started follow-up visits every two months. Five months after radiotherapy she underwent a computerised tomography scan of the abdomen, which revealed a 4-cm mass in the liver and a second-line chemotherapy with gemcitabine was given. A positron emission tomography after completion of the third cycle of chemotherapy with gemcitabine showed the presence of new lesions into the liver and an elevation of CA125 level at 458 U/ml was also observed. Thus the patient received another line of chemotherapy with topotecan, interrupted after three cycles due to progressive disease from the evidence of peritoneal metastases at the tomography scan of the abdomen and pelvis and a new increase in CA125 level to 1482 U/ml. She interrupted the medical treatment and an ileostomy due to bowel occlusion was lately performed. The patient died of progressive disease three months later.
Discussion
The most frequent sites of implantation of ectopic endometrium are ovaries, Douglas pouch, and broad and uterosacral ligaments (Schwartzwald et al. 1992). The frequency of urinary tract involvement is a rare event and was first reported by Judd in 1921; its incidence is thought to be around 1%, and 84% of all cases involve the bladder (Shook and Nyberg 1988).
Case presentation
A 38-year-old Caucasian woman was admitted in our department for pelvic pain, uterine fibroids and infertility. She presented with microscopic haematuria, dysuria and moderate pelvic pain that started approximately before two months. Before 3 years the patient underwent a laparotomy with bilateral excision of endometriomas and myomectomy. The physical examination revealed only mild tenderness in the pubic region, but no masses were palpable during transvaginal examination. Serum level of CA125 was increased (45.5 U/ml, normal range: 0–35 U/ml), while other tumour markers (CA19.9, CA15.3, CEA and AFP) were normal. Transvaginal ultrasound scans showed an irregular, solid, 7-cm large mass, possibly growing from the posterior wall
Figure 1. Pathological figure revealed a surrounding endometriosis area in which we observed a tumour involving the bladder wall with mucosal saving.
ISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20
Vaginal teratoma: A case report and review of the literature F. Vural, B. Vural & N. Paksoy To cite this article: F. Vural, B. Vural & N. Paksoy (2015) Vaginal teratoma: A case report and review of the literature, Journal of Obstetrics and Gynaecology, 35:7, 757-758, DOI: 10.3109/01443615.2015.1004525 To link to this article: http://dx.doi.org/10.3109/01443615.2015.1004525
Published online: 11 Feb 2015.
Submit your article to this journal
Article views: 20
View related articles
View Crossmark data
Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=ijog20 Download by: [University of Otago]
Date: 19 October 2015, At: 08:11
Gynaecology Case Reports 757
Vaginal teratoma: A case report and review of the literature F. Vural1, B. Vural2 & N. Paksoy3 1Department of Obstetrics and Gynecology, Haydarpaşa Numune
Teaching Hospital, Istanbul, Turkey, 2Department of Obstetrics and Gynecology, Kocaeli University Faculty of Medicine, Kocaeli, Turkey, and 3Department of Pathology, Kocaeli University Faculty of Medicine, Kocaeli, Turkey DOI: 10.3109/01443615.2015.1004525 Correspondence: Fisun Vural, M.D., Orhan mah. Bağtepe Sok. Erenkaya park Evleri A2/2 Bağçeşme/Kocaeli, Turkey. E-mail: [email protected]
Downloaded by [University of Otago] at 08:11 19 October 2015
Introduction
Dermoid cysts (benign cystic teratoma) are benign germ-cell tumours that contain well-differentiated derivatives of all three germ layers (Sharma et al. 2012). They usually occur along the line of embryonic fusion over the head and neck region, the mediastinum and the presacral area and are present at birth or develop in early childhood. Although most teratomas occur in the midline, they may present atypically as vaginal dermoid cysts (Sharma et al. 2012; Siu et al, 2003). A true cutaneous dermoid cyst is formed by ectodermal cells buried along the lines of closure of embryonic clefts and sinuses by skin fusion during the embryonic stage, these cells are able to transform into hair, skin and appendages. If dermoid cyst contains mesodermal cells, bone, teeth and cartilage may develop. If teeth are found in the vaginal dermoid cyst, it is known as ‘vagina dentata’ (Sharma et al. 2012; Siu et al, 2003). Mention of vagina dentata is found in the creation myths and folk stories of many cultures such as Native Americans in North America and different populations in Russia, Japan, India, Samoa and New Zealand. After the 19th century, these stories were interpreted as the ‘castration anxiety of man’ (Jackson, 1971). Although vagina dentata is a universal symbol of men’s fear of women, vaginal dermoid cyst is quite rare. To the best of our knowledge, there are 7 cases of vaginal dermoid cysts published thus far in the English literature (Sharma et al. 2012). We present a case of a vaginal dermoid cyst along with a review of the literature.
Case report Clinical history A 15-year-old girl presented with a protruding mass from the hymenal ring. Her height was 160 cm and she weighed 40 kg. She
Figure 1. Microscopic appearance of vaginal teratoma. The dilated mature glandular tissue and mature cartilage tissue. (Figure 1. haematoxylin and eosin; 125).
had a history of regular menstrual cycles, was Tanner stage 5 in breast development and had a normal body hair growth pattern. There was no history of in utero exposure to diethylstilbestrol, radiotherapy or any medical disorders. The ultrasonographic findings of the pelvis were normal: uterus (60 55 45 mm), right ovary (25 20 20 mm) and left ovary (20 18 15 mm) with normal follicular appearance. Gynaecological examination revealed both soft and semisolid, polypoid mobile structures with a smooth surface protruding through the introitus (hymenal ring). The polypoid tissue measured 5 4 3 cm with a stalk that was 0.5 cm in width attached to the lower lateral wall of the vaginal submucosal tissue. Complete excision of the mass was performed under local anaesthesia. The patient’s post-operative course was uneventful.
Histopathological findings Macroscopically, a polypoid structure measuring 5 4 3 cm was described with necrotic margins. The cut surface demonstrated three well-demarcated areas of cartilage tissue embedded within the structure. The histopathological examination revealed a polypoid tissue covered by squamous epithelium with some reactive changes. Subepithelial connective tissue contained irregular-shaped, thin-walled, dilated glandular structures and a few islands of mature cartilage tissue. There were no other adult tissues. No immature elements were also seen. The diagnosis was a mature teratoma, shown in Figure 1.
Discussion
Vaginal cysts are not uncommon findings in clinical practice – epidermal inclusion cysts, Gardner’s duct cysts and Bartholin’s duct cysts are common types. However, dermoid cyst, ectopic ureterocele and paraurethral cyst is rare types (Siu et al, 2003). Dermoid cysts are a special form of mature teratoma and they represent the most common benign germ-cell tumour and the most common neoplasms of the ovary in young women (Sharma et al. 2012). Dermoid cysts are usually present at birth and continue to grow in size with age. The cyst is similar to those present on skin tissue and can contain fat, and occasionally hair, bone, nails, teeth, eyes, cartilage and thyroid tissue. On occasion, teeth can be found within the vaginal dermoid cyst, which is why they are called ‘vagina dentata’ (Sharma et al. 2012; Siu et al, 2003). Although vagina dentata appears in the creation of myths and folkloric stories of many cultures (Jackson, 1971), a dermoid cyst of the vagina is quite rare. Vaginal dermoid cysts were first described in 1899 by Stokes, who reported a 44-year-old woman who underwent removal of a 1-cm cyst located within the hymen. The cyst contained numerous sebaceous glands and a few hair follicles (Stokes 1899). Curtis (1913) described an ulcerated orange-sized necrotic cyst containing hair and sebaceous materials in the vaginal mucosa. In another case report, a 4-inch cyst was expelled from a woman’s vagina following delivery of her second child. The cyst contained thick sebaceous material with matted hair and had been attached to the vaginal wall by a narrow stalk (Johnston 1939). In still another report, excision of a painful right vaginal wall cyst revealed it to be a dermoid cyst confirmed by histopathological examination (Hirose et al, 1991). Subsequently, Siu et al (2003), AlShahrani and Heywood (2006) and Sharma et al. (2012) reported three cases of paravaginal dermoid cysts. Our case differed from the others in that 1) the girl was an adolescent; 2) the mass was a smooth-based multiple polypoid mass with a stalk and 3) the cyst was not located in the paravaginal space but rather protruded from the hymen. Siu et al (2003) noted that cutaneous dermoid cysts and epidermal inclusion cysts of the vagina had similar characteristics. During the repair of an episiotomy, skin appendages may become buried in the wound leading to features that are consistent with a dermoid cyst. However, this latter case involved a 15-year-old adolescent girl with no history of sexual intercourse. Thus, the aetiology of vaginal dermoids remains to be elucidated. Vaginal teratoma is a rare and benign condition that can be diagnosed by histopathological examination and treated with surgical excision. Declaration of interests: The authors report no declarations of interest. The authors alone are responsible for the content and writing of the paper.
758 Gynaecology Case Reports
References
Downloaded by [University of Otago] at 08:11 19 October 2015
Al-Shahrani M, Heywood M. 2006. A rare case of vaginal dermoid cyst: a case report and review of literature. Biomedical Research 17:95–97. Curtis AH. 1913. Transactions of societies. Chicago Gynecological Society. Surgery, Gynecology and Obstetrics 16:715. Hirose R, Imai A, Kondo H, Itoh K, Tamaya T. 1991. A dermoid cyst of the paravaginal space. Archives of Gynecology and Obstetrics. 249:39–41. Jackson B. 1971. Vagina dentata and cystic teratoma. Journal of American Folklore 84:341–342. Johnston HW. 1939. A dermoid cyst of the vagina complicated by pregnancy. Canadian Medical Association Journal 41:386. Sharma R, Rao KA, Nagraj S. 2012. A rare case of vaginal dermoid cyst: a case report and review of literature. International Journal of Infertility and Fetal Medicine 3:102–104. Siu SS, Tam WH, To KF, Yuen PM. 2003. Is vaginal dermoid cyst a rare occurrence or a misnomer? A case report and review of the literature. Ultrasound in Obstetrics & Gynecology 21:404–406. Stokes JE. 1899. The etiology and structure of true vaginal cysts. Johns Hopkins Hospital Reports 7:109–136.
A rare case of primary clear-cell adenocarcinoma of the bladder arising from bladder endometriosis V. Loizzi1,2, G. Cormio1,2, L. Leone1, M. Falagario1, S. Longo3, L. Resta4 & L. Selvaggi1 1Department of Obstetrics, Gynecology and Neonatology, University
of Bari, Bari, Italy, 2Division of Gynecology Oncology, IRCCS Bari, University of Bari, Bari, Italy, 3Department of Internal Medicine, University of Bari, Bari, Italy, and 4Department of Pathology, University of Bari, Bari, Italy DOI: 10.3109/01443615.2015.1004526 Correspondence: Vera Loizzi, M.D., Aggregate Professor, Department of Obstetrics, Gynecology and Neonatology, University of Bari, Piazza Giulio Cesare 11, 70124 Bari, Italy. E-mail: [email protected]
Introduction
Endometriosis is a common benign gynaecological condition affecting 5–15% of women in their reproductive age and only 3–5% of post-menopausal women, in which endometrial tissue grows outside uterine cavity, leading to pelvic pain and infertility (Vignali et al. 2002). Even if endometriosis is a benign condition, it can spread to other organs by invading and disrupting their tissues, sharing these characteristics with invasive cancers. It is well known that ovarian cancer, especially endometrioid and clear cell types, can be the result of a malignant transformation of foci of endometriosis (Bacci et al. 2009), but even if extra-gonadic endometriosis malignant transformation was considered an exceptional event, recent studies suggested that it can also be associated with other kinds of cancer (Ness 2003; Olson et al. 2002; Scully et al. 1966). Primary clear-cell bladder adenocarcinoma is a very rare tumour, affecting mostly women; it can be associated with bladder endometriosis suggesting a mullerian origin of this rare cancer (Lu et al. 2012).
of the bladder. The patient underwent a surgery with a laparotomic approach. An extended pelvic disease was observed. Only uterine fundus was visible because bladder was completely adherent to its anterior wall, and posteriorly adnexa were fixed to posterior uterine wall. An approximately 8-cm mass involving the whole bladder vault was evaluated. A partial cystectomy with removal of left ovarian cyst was performed. The final pathology report revealed a surrounding endometriosis zone in which we observed a tumour involving the bladder wall with mucosal saving (Figure 1). This tumour is composed of an aggregate of glandular structures with lining monofilament epithelial that in some areas appear flattened or cubical, whereas in others it is composed of micropapillary protrusions showing an axis more often hyaline and prominent vesicular nuclei on the epithelium and a large nucleolus. There were sporadic calcified concretions such as psammoma bodies. The periodic acid–Schiff reaction with diastase showed occasional presence of intracytoplasmic glycogen. According to Sampson’s criteria, diagnosis of clear-cell adenocarcinoma (CCA) of the bladder arisen from bladder endometriosis was made. The absence of ovarian and endometrial involvement leads us to believe that neoplasm should be a primary tumour in the bladder wall. The patient underwent 4 cycles of chemotherapy with paclitaxel, cisplatin and epirubicin (TEP). Afterwards a laparotomy with bilateral pelvic lymphadenectomy, radical Piver type II hysterectomy and bilateral salpingo-oophorectomy was performed. The pathology report revealed the presence of CCA in the pelvic lymph nodes, uterine serosa and ovaries. Other two cycles of TEP and external pelvic radiotherapy, with a total dose of 4500 cGy, were then given to the patient. The patient started follow-up visits every two months. Five months after radiotherapy she underwent a computerised tomography scan of the abdomen, which revealed a 4-cm mass in the liver and a second-line chemotherapy with gemcitabine was given. A positron emission tomography after completion of the third cycle of chemotherapy with gemcitabine showed the presence of new lesions into the liver and an elevation of CA125 level at 458 U/ml was also observed. Thus the patient received another line of chemotherapy with topotecan, interrupted after three cycles due to progressive disease from the evidence of peritoneal metastases at the tomography scan of the abdomen and pelvis and a new increase in CA125 level to 1482 U/ml. She interrupted the medical treatment and an ileostomy due to bowel occlusion was lately performed. The patient died of progressive disease three months later.
Discussion
The most frequent sites of implantation of ectopic endometrium are ovaries, Douglas pouch, and broad and uterosacral ligaments (Schwartzwald et al. 1992). The frequency of urinary tract involvement is a rare event and was first reported by Judd in 1921; its incidence is thought to be around 1%, and 84% of all cases involve the bladder (Shook and Nyberg 1988).
Case presentation
A 38-year-old Caucasian woman was admitted in our department for pelvic pain, uterine fibroids and infertility. She presented with microscopic haematuria, dysuria and moderate pelvic pain that started approximately before two months. Before 3 years the patient underwent a laparotomy with bilateral excision of endometriomas and myomectomy. The physical examination revealed only mild tenderness in the pubic region, but no masses were palpable during transvaginal examination. Serum level of CA125 was increased (45.5 U/ml, normal range: 0–35 U/ml), while other tumour markers (CA19.9, CA15.3, CEA and AFP) were normal. Transvaginal ultrasound scans showed an irregular, solid, 7-cm large mass, possibly growing from the posterior wall
Figure 1. Pathological figure revealed a surrounding endometriosis area in which we observed a tumour involving the bladder wall with mucosal saving.
