Gynaecology Case Reports 215
Figure 1. Pathological and histological examination of an ovary from a subject presenting for haemangioma with hyperandrogenism. (a) A cross-sectional view of the right ovary after fixation shows a haemorrhagic mass approximately 1 cm in diameter defined by a yellow area of tissue. (b,c) Haematoxylin and eosin staining of the sections obtained from the ovary shown in (a). Capillary haemangioma was characterised by numerous small spaces filled with red blood cells (b, ⫻ 40 magnification). Ovarian stromal cells showed extensive luteinisation (c, ⫻ 100 magnification).
J Obstet Gynaecol Downloaded from informahealthcare.com by Selcuk Universitesi on 02/01/15 For personal use only.
Introduction Vascular tumours of the ovary are extremely rare, with approximately 50 cases of ovarian haemangioma reported so far (Uppal et al. 2004). Ovarian haemangiomas are ‘non-functional’ neoplasms and seldom cause hyperandrogenism. We present a case of ovarian haemangioma characterised by male pattern baldness and an elevated testosterone level.
Case report A 66-year-old Japanese woman, postmenopausal for 21 years, was followed by her primary care physician for hypertension. She was referred to our department because of beard growth, which spanned a period of 2 years. Her frontal scalp hair was thin. Ultrasonographic examination revealed an enlarged right ovary (3.0 ⫻ 2.1 ⫻ 2.8 cm) with increased vascularisation noted by Doppler ultrasound, which was confirmed by computerised tomography (CT) and magnetic imaging resonance (MRI) scans. Preoperative serum hormone levels were as follows: follicle stimulating hormone (FSH) 10.95 mIU/ ml; luteinising hormone (LH) 3.65 mIU/ml; oestradiol 55 pg/ml (normal range for menopausal women, 0–21 pg/ml); and testosterone 4.93 ng/ml (normal range, 0.13–0.69 ng/ml). Serum levels of 17α-hydroxypregnenolone, dehydroepiandrostenedione sulphate and 11-hydroxycorticosteroid were normal. Also, oral challenge with progesterone did not result in uterine bleeding. Laparoscopy revealed the right ovary to be slightly enlarged. A bilateral salpingooophorectomy was performed. A pathological investigation showed the presence of a dark red spongy haemorrhagic mass surrounded by a bright-coloured area (Figure 1a). Subsequent histological analysis of frozen cross-sections by routine haematoxylin and eosin (H&E) staining confirmed the presence of a haemangioma and fatty mass without cytological atypia. The haemangioma was characterised by small, thin-walled, capillary-like spaces, lined with a monolayer of endothelial cells and filled with red blood cells (Figure 1b). Mitotic activity was not noted, and atypical cells were not observed. Reinke’s crystals were also not seen. Ovarian stromal cells had undergone extensive luteinisation (Figure 1c), and the cortex was attenuated but intact. The final histological diagnosis was capillary haemangioma with stromal luteinisation. The left ovary was normal. Serum concentrations of gonadotropins and sex steroids returned to normal levels and male pattern baldness was alleviated after surgery. There were no changes in the amount of the antihypertensive drugs she needed.
Discussion Haemangiomas are benign vascular tumours rarely found in the ovary, with approximately 50 cases of ovarian haemangioma reported so far (Uppal et al. 2004). They are usually small ‘non-functional’ neoplasms and most are found accidentally during surgery. To the best of our knowledge, there is only one reported case of ovarian haemangioma with hyperandrogenism (Gücer et al. 2004). Clinical virilisation has also been shown to associate with many benign, malignant, primary or metastatic ‘non-functional’ ovarian neoplasms (Nezhat et al. 2002). Moreover, luteinisation of the ovarian stroma in areas adjacent to non-endocrinological neoplasms has been reported. Indeed, luteinisation of stromal tissue was apparent in our case of ovarian haemangioma. It is well known that luteinised stromal cells produce steroids, mainly androgens (Gücer et al. 2004).
The mechanism behind stromal luteinisation is unclear. It is possible that the neoplasm produces a mechanical effect on the stroma, similar to an expanding follicle (Nezhat et al. 2002). It should be noted, however, that not all ovarian tumours induce stromal luteinisation. One theory is that some tumours produce a growth promoting factor that triggers stromal hyperplasia (Nezhat et al. 2002). In summary, we have presented a second case of ovarian haemangioma with virilisation. We conclude that the ovarian stroma contained luteinised cells, resulting in the production of androgens and in the clinical manifestations present in this patient. When we find ovarian masses with virilisation, we should think about the possibility of non-functional tumours other than hormone-producing tumours. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Gücer F, Ozyilmaz F, Balkanli-Kaplan P et al. 2004. Ovarian hemangioma presenting with hyperandrogenism and endometrial cancer: a case report. Gynecologic Oncology 94:821–824. Nezhat F, Slomovitz BM, Saiz AD et al. 2002. Ovarian mucinous cystadenocarcinoma with virilization. Gynecologic Oncology 84:468–472. Uppal S, Heller DS, Majmudar B. 2004. Ovarian hemangioma – report of three cases and review of the literature. Archives of Gynecology and Obstetrics 270:1–5.
Ovarian sclerosing stromal tumour with elevated CA19-9 levels A. Karabulut1, N. Karabulut2 & M. Akbulut3 Departments of 1Obstetrics and Gynecology, 2Radiology and 3Pathology, Pamukkale University Medical School, Denizli, Turkey DOI: 10.3109/01443615.2014.940289 Correspondence: A. Karabulut, Onelya Evleri, A-4 Yenişehir 20125 Denizli, Turkey. E-mail: [email protected]
Introduction Sclerosing stromal tumour (SST) of the ovary is a rare benign tumour of the ovarian stroma, first described in 1973 (Chalvardjian and Scully 1973). Although it is classified within the sex cord stromal tumour category, it shows distinctive clinical and pathological features (Chalvardjian and Scully 1973). It is predominantly seen in the second and third decades of life and is usually hormonally inactive (Chalvardjian and Scully 1973; Marelli et al. 1998). However, rarely it may show androgenic activity (Park et al. 2011). Diagnosis is often made by pathological examination. Tumour markers usually remain normal in these patients. We present here a case of SST of the ovary with elevated CA19-9 levels.
J Obstet Gynaecol Downloaded from informahealthcare.com by Selcuk Universitesi on 02/01/15 For personal use only.
216
Gynaecology Case Reports
Figure 1. Contrast-enhanced CT showing a well-defined right ovarian mass with an avidly enhancing peripheral solid region and a hypodense centre due to cystic-necrotic components.
Case report A 20-year-old virgin female was admitted with the complaint of intermittent abdominal pain 3–4 times in a week that was hard to localise. She also had menstrual irregularity over the last few months. On sonographic examination, a 5 ⫻ 6 cm semisolid mass was detected on the right adnexum. CA19-9 level was 66.3 IU/ml, other tumour markers and laboratory values were within normal limits. No abnormality was detected in gastrointestinal screening. Computed tomography (CT) of the pelvis revealed a solid, well-defined right ovarian mass measuring 53 ⫻ 38 mm. The periphery of the mass enhanced avidly after i.v. contrast administration, while central cystic-necrotic portions remained hypodense (Figure 1). Radiological features were consistent with benign ovarian mass, with a tentative diagnosis of SST. Laparotomy depicted a solid 6 ⫻ 5 ⫻ 3 cm sized mass in the right ovary. A right salpingooophorectomy was performed along with peritoneal washing. The contralateral ovary and uterus were normal in appearance. There was no evidence of ascites or, pelvic or intra-abdominal lymphadenopathy. Pathological examination revealed sclerosing stromal tumour of the ovary. The cut surface was lobulated with solid areas, yellowishwhite in colour, and there were central areas of oedema. Microscopic examination revealed a pseudo-lobular pattern in which cellular nodules were separated by areas of densely collagenous or oedematous connective tissue. The nodules were an admixture of fibroblasts and rounded vacuolated cells, with a prominent nucleus. No cytological evidence of malignancy was seen. Immunohistochemical staining for CD34 demonstrated extensive vascularity, and staining for inhibin and smooth muscle actin showed a positive reaction, supporting the diagnosis of sclerosing stromal tumour of the ovary. The postoperative period was uneventful, and CA19-9 levels returned to normal on the 20th postoperative day.
Discussion
Sclerosing stromal tumour is a rare neoplasm comprising ⬍ 5% of sex cord stromal tumours (Outwater et al. 1998). The most common presenting symptoms are menstrual irregularities, abdominal discomfort and pain. In our patient, the major symptoms were as described; abdominal pain and menstrual irregularity. Sclerosing stromal tumour may rarely show hormonal activity. Androgen secretion resulting in virilisation and elevated CA125 levels were described previously (Terauchi et al. 2005; Park et al. 2011). However, elevated CA19-9 levels in SST has not been reported previously in the literature.
CA19-9 is a carbohydrate determinant recognised by a monoclonal antibody originally developed against a human colon carcinoma cell line (Trapé et al. 2011). It is usually elevated in mucin secreting tumours (Rhodes 1999). However, we could not detect mucin production in our case. CA19-9 is considered the most valuable serum test used in the diagnosis and management of pancreatic and gastrointestinal malignancies (Steinberg 1990). For this reason, we did gastrointestinal screening in our patient. A CA19-9 value of ⬎ 1,000 U/ml usually indicates a digestive cancer and has been reported to have a specificity over 99% for pancreatic cancer (Steinberg 1990). Moderately elevated serum levels of CA19-9 can be detected during benign diseases of the liver, pancreas and biliary tract (Steinberg 1990). Radiographic imaging may be instrumental in cases with elevated tumour markers. Sonography, CT or magnetic resonance imaging (MRI) not only detect and localise the mass but also provide suggestive features for the nature, such as solid, cystic, haemorrhagic or complex (Matsubayashi et al. 1999; Terauchi et al. 2005). Areas of calcification or lipid tissue can also be readily detected by CT. Furthermore, contrast enhancement pattern helps distinguish benign versus malignant tumours, and sometimes it may indicate a particular tumour. The well-defined contour of the ovarian mass and absence of ascites and lymphadenopathy were suggestive for a benign-borderline mass, and intense peripheral enhancement was consistent with SST. Thick capsulated mass showing peripheral contrast enhancement with centripetal progression has consistently been described in cases with SST (Matsubayashi et al. 1999; Kim et al. 2003). The rich vascular network of the peripheral part of the tumour is most likely responsible for the peripheral contrast enhancement on CT and MRI, and the oedematous stroma of the tumour midportion explains the mild and late enhancement that is typical of stromal tissue (Kim et al. 2003). In conclusion, SST is a benign tumour of the ovary with characteristic clinical, pathological and radiological features. As it rarely accompanies elevated CA19-9 and CA125 levels, clinical diagnosis would be complicated. Suggestive imaging features may help the gynaecologist undertake appropriate surgical techniques in a young women presenting with an ovarian mass. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Chalvardjian A, Scully RE. 1973. Sclerosing stromal tumors of the ovary. Cancer 31:664–670. Kim JY, Jung KJ, Chung DS et al. 2003. Sclerosing stromal tumor of the ovary: MR-pathologic correlation in three cases. Korean Journal of Radiology 4: 194–199. Marelli G, Carinelli S, Mariani A et al. 1998. Sclerosing stromal tumor of the ovary. Report of eight cases and review of the literature. European Journal of Obstetrics, Gynecology, and Reproductive Biology 76:85–89. Matsubayashi R, Matsuo Y, Doi J et al. 1999. Sclerosing stromal tumor of the ovary: radiologic findings. European Radiology 9:1335–1338. Outwater EK, Wagner BJ, Mannion C et al. 1998. Sex cord-stromal and steroid cell tumors of the ovary. Radiographics 18:1523–1546. Park SM, Kim YN, Woo YJ et al. 2011. A sclerosing stromal tumor of the ovary with masculinization in a premenarchal girl. Korean Journal of Pediatrics 54:224–227. Rhodes JM. 1999. Usefulness of novel tumour markers. Annals of Oncology 10(Suppl 4):118–121. Steinberg WM. 1990. The clinical utility of the CA 19-9 tumor-associated antigen. American Journal of Gastroenterology 85:350–355. Terauchi F, Onodera T, Nagashima T et al. 2005. Sclerosing stromal tumor of the ovary with elevated CA125. Journal of Obstetrics and Gynaecology Research 31:432–435. Trapé J, Filella X, Alsina-Donadeu M et al. 2011. Increased plasma concentrations of tumour markers in the absence of neoplasia. Clinical Chemistry and Laboratory Medicine 49:1605–1620.
Figure 1. Pathological and histological examination of an ovary from a subject presenting for haemangioma with hyperandrogenism. (a) A cross-sectional view of the right ovary after fixation shows a haemorrhagic mass approximately 1 cm in diameter defined by a yellow area of tissue. (b,c) Haematoxylin and eosin staining of the sections obtained from the ovary shown in (a). Capillary haemangioma was characterised by numerous small spaces filled with red blood cells (b, ⫻ 40 magnification). Ovarian stromal cells showed extensive luteinisation (c, ⫻ 100 magnification).
J Obstet Gynaecol Downloaded from informahealthcare.com by Selcuk Universitesi on 02/01/15 For personal use only.
Introduction Vascular tumours of the ovary are extremely rare, with approximately 50 cases of ovarian haemangioma reported so far (Uppal et al. 2004). Ovarian haemangiomas are ‘non-functional’ neoplasms and seldom cause hyperandrogenism. We present a case of ovarian haemangioma characterised by male pattern baldness and an elevated testosterone level.
Case report A 66-year-old Japanese woman, postmenopausal for 21 years, was followed by her primary care physician for hypertension. She was referred to our department because of beard growth, which spanned a period of 2 years. Her frontal scalp hair was thin. Ultrasonographic examination revealed an enlarged right ovary (3.0 ⫻ 2.1 ⫻ 2.8 cm) with increased vascularisation noted by Doppler ultrasound, which was confirmed by computerised tomography (CT) and magnetic imaging resonance (MRI) scans. Preoperative serum hormone levels were as follows: follicle stimulating hormone (FSH) 10.95 mIU/ ml; luteinising hormone (LH) 3.65 mIU/ml; oestradiol 55 pg/ml (normal range for menopausal women, 0–21 pg/ml); and testosterone 4.93 ng/ml (normal range, 0.13–0.69 ng/ml). Serum levels of 17α-hydroxypregnenolone, dehydroepiandrostenedione sulphate and 11-hydroxycorticosteroid were normal. Also, oral challenge with progesterone did not result in uterine bleeding. Laparoscopy revealed the right ovary to be slightly enlarged. A bilateral salpingooophorectomy was performed. A pathological investigation showed the presence of a dark red spongy haemorrhagic mass surrounded by a bright-coloured area (Figure 1a). Subsequent histological analysis of frozen cross-sections by routine haematoxylin and eosin (H&E) staining confirmed the presence of a haemangioma and fatty mass without cytological atypia. The haemangioma was characterised by small, thin-walled, capillary-like spaces, lined with a monolayer of endothelial cells and filled with red blood cells (Figure 1b). Mitotic activity was not noted, and atypical cells were not observed. Reinke’s crystals were also not seen. Ovarian stromal cells had undergone extensive luteinisation (Figure 1c), and the cortex was attenuated but intact. The final histological diagnosis was capillary haemangioma with stromal luteinisation. The left ovary was normal. Serum concentrations of gonadotropins and sex steroids returned to normal levels and male pattern baldness was alleviated after surgery. There were no changes in the amount of the antihypertensive drugs she needed.
Discussion Haemangiomas are benign vascular tumours rarely found in the ovary, with approximately 50 cases of ovarian haemangioma reported so far (Uppal et al. 2004). They are usually small ‘non-functional’ neoplasms and most are found accidentally during surgery. To the best of our knowledge, there is only one reported case of ovarian haemangioma with hyperandrogenism (Gücer et al. 2004). Clinical virilisation has also been shown to associate with many benign, malignant, primary or metastatic ‘non-functional’ ovarian neoplasms (Nezhat et al. 2002). Moreover, luteinisation of the ovarian stroma in areas adjacent to non-endocrinological neoplasms has been reported. Indeed, luteinisation of stromal tissue was apparent in our case of ovarian haemangioma. It is well known that luteinised stromal cells produce steroids, mainly androgens (Gücer et al. 2004).
The mechanism behind stromal luteinisation is unclear. It is possible that the neoplasm produces a mechanical effect on the stroma, similar to an expanding follicle (Nezhat et al. 2002). It should be noted, however, that not all ovarian tumours induce stromal luteinisation. One theory is that some tumours produce a growth promoting factor that triggers stromal hyperplasia (Nezhat et al. 2002). In summary, we have presented a second case of ovarian haemangioma with virilisation. We conclude that the ovarian stroma contained luteinised cells, resulting in the production of androgens and in the clinical manifestations present in this patient. When we find ovarian masses with virilisation, we should think about the possibility of non-functional tumours other than hormone-producing tumours. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Gücer F, Ozyilmaz F, Balkanli-Kaplan P et al. 2004. Ovarian hemangioma presenting with hyperandrogenism and endometrial cancer: a case report. Gynecologic Oncology 94:821–824. Nezhat F, Slomovitz BM, Saiz AD et al. 2002. Ovarian mucinous cystadenocarcinoma with virilization. Gynecologic Oncology 84:468–472. Uppal S, Heller DS, Majmudar B. 2004. Ovarian hemangioma – report of three cases and review of the literature. Archives of Gynecology and Obstetrics 270:1–5.
Ovarian sclerosing stromal tumour with elevated CA19-9 levels A. Karabulut1, N. Karabulut2 & M. Akbulut3 Departments of 1Obstetrics and Gynecology, 2Radiology and 3Pathology, Pamukkale University Medical School, Denizli, Turkey DOI: 10.3109/01443615.2014.940289 Correspondence: A. Karabulut, Onelya Evleri, A-4 Yenişehir 20125 Denizli, Turkey. E-mail: [email protected]
Introduction Sclerosing stromal tumour (SST) of the ovary is a rare benign tumour of the ovarian stroma, first described in 1973 (Chalvardjian and Scully 1973). Although it is classified within the sex cord stromal tumour category, it shows distinctive clinical and pathological features (Chalvardjian and Scully 1973). It is predominantly seen in the second and third decades of life and is usually hormonally inactive (Chalvardjian and Scully 1973; Marelli et al. 1998). However, rarely it may show androgenic activity (Park et al. 2011). Diagnosis is often made by pathological examination. Tumour markers usually remain normal in these patients. We present here a case of SST of the ovary with elevated CA19-9 levels.
J Obstet Gynaecol Downloaded from informahealthcare.com by Selcuk Universitesi on 02/01/15 For personal use only.
216
Gynaecology Case Reports
Figure 1. Contrast-enhanced CT showing a well-defined right ovarian mass with an avidly enhancing peripheral solid region and a hypodense centre due to cystic-necrotic components.
Case report A 20-year-old virgin female was admitted with the complaint of intermittent abdominal pain 3–4 times in a week that was hard to localise. She also had menstrual irregularity over the last few months. On sonographic examination, a 5 ⫻ 6 cm semisolid mass was detected on the right adnexum. CA19-9 level was 66.3 IU/ml, other tumour markers and laboratory values were within normal limits. No abnormality was detected in gastrointestinal screening. Computed tomography (CT) of the pelvis revealed a solid, well-defined right ovarian mass measuring 53 ⫻ 38 mm. The periphery of the mass enhanced avidly after i.v. contrast administration, while central cystic-necrotic portions remained hypodense (Figure 1). Radiological features were consistent with benign ovarian mass, with a tentative diagnosis of SST. Laparotomy depicted a solid 6 ⫻ 5 ⫻ 3 cm sized mass in the right ovary. A right salpingooophorectomy was performed along with peritoneal washing. The contralateral ovary and uterus were normal in appearance. There was no evidence of ascites or, pelvic or intra-abdominal lymphadenopathy. Pathological examination revealed sclerosing stromal tumour of the ovary. The cut surface was lobulated with solid areas, yellowishwhite in colour, and there were central areas of oedema. Microscopic examination revealed a pseudo-lobular pattern in which cellular nodules were separated by areas of densely collagenous or oedematous connective tissue. The nodules were an admixture of fibroblasts and rounded vacuolated cells, with a prominent nucleus. No cytological evidence of malignancy was seen. Immunohistochemical staining for CD34 demonstrated extensive vascularity, and staining for inhibin and smooth muscle actin showed a positive reaction, supporting the diagnosis of sclerosing stromal tumour of the ovary. The postoperative period was uneventful, and CA19-9 levels returned to normal on the 20th postoperative day.
Discussion
Sclerosing stromal tumour is a rare neoplasm comprising ⬍ 5% of sex cord stromal tumours (Outwater et al. 1998). The most common presenting symptoms are menstrual irregularities, abdominal discomfort and pain. In our patient, the major symptoms were as described; abdominal pain and menstrual irregularity. Sclerosing stromal tumour may rarely show hormonal activity. Androgen secretion resulting in virilisation and elevated CA125 levels were described previously (Terauchi et al. 2005; Park et al. 2011). However, elevated CA19-9 levels in SST has not been reported previously in the literature.
CA19-9 is a carbohydrate determinant recognised by a monoclonal antibody originally developed against a human colon carcinoma cell line (Trapé et al. 2011). It is usually elevated in mucin secreting tumours (Rhodes 1999). However, we could not detect mucin production in our case. CA19-9 is considered the most valuable serum test used in the diagnosis and management of pancreatic and gastrointestinal malignancies (Steinberg 1990). For this reason, we did gastrointestinal screening in our patient. A CA19-9 value of ⬎ 1,000 U/ml usually indicates a digestive cancer and has been reported to have a specificity over 99% for pancreatic cancer (Steinberg 1990). Moderately elevated serum levels of CA19-9 can be detected during benign diseases of the liver, pancreas and biliary tract (Steinberg 1990). Radiographic imaging may be instrumental in cases with elevated tumour markers. Sonography, CT or magnetic resonance imaging (MRI) not only detect and localise the mass but also provide suggestive features for the nature, such as solid, cystic, haemorrhagic or complex (Matsubayashi et al. 1999; Terauchi et al. 2005). Areas of calcification or lipid tissue can also be readily detected by CT. Furthermore, contrast enhancement pattern helps distinguish benign versus malignant tumours, and sometimes it may indicate a particular tumour. The well-defined contour of the ovarian mass and absence of ascites and lymphadenopathy were suggestive for a benign-borderline mass, and intense peripheral enhancement was consistent with SST. Thick capsulated mass showing peripheral contrast enhancement with centripetal progression has consistently been described in cases with SST (Matsubayashi et al. 1999; Kim et al. 2003). The rich vascular network of the peripheral part of the tumour is most likely responsible for the peripheral contrast enhancement on CT and MRI, and the oedematous stroma of the tumour midportion explains the mild and late enhancement that is typical of stromal tissue (Kim et al. 2003). In conclusion, SST is a benign tumour of the ovary with characteristic clinical, pathological and radiological features. As it rarely accompanies elevated CA19-9 and CA125 levels, clinical diagnosis would be complicated. Suggestive imaging features may help the gynaecologist undertake appropriate surgical techniques in a young women presenting with an ovarian mass. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Chalvardjian A, Scully RE. 1973. Sclerosing stromal tumors of the ovary. Cancer 31:664–670. Kim JY, Jung KJ, Chung DS et al. 2003. Sclerosing stromal tumor of the ovary: MR-pathologic correlation in three cases. Korean Journal of Radiology 4: 194–199. Marelli G, Carinelli S, Mariani A et al. 1998. Sclerosing stromal tumor of the ovary. Report of eight cases and review of the literature. European Journal of Obstetrics, Gynecology, and Reproductive Biology 76:85–89. Matsubayashi R, Matsuo Y, Doi J et al. 1999. Sclerosing stromal tumor of the ovary: radiologic findings. European Radiology 9:1335–1338. Outwater EK, Wagner BJ, Mannion C et al. 1998. Sex cord-stromal and steroid cell tumors of the ovary. Radiographics 18:1523–1546. Park SM, Kim YN, Woo YJ et al. 2011. A sclerosing stromal tumor of the ovary with masculinization in a premenarchal girl. Korean Journal of Pediatrics 54:224–227. Rhodes JM. 1999. Usefulness of novel tumour markers. Annals of Oncology 10(Suppl 4):118–121. Steinberg WM. 1990. The clinical utility of the CA 19-9 tumor-associated antigen. American Journal of Gastroenterology 85:350–355. Terauchi F, Onodera T, Nagashima T et al. 2005. Sclerosing stromal tumor of the ovary with elevated CA125. Journal of Obstetrics and Gynaecology Research 31:432–435. Trapé J, Filella X, Alsina-Donadeu M et al. 2011. Increased plasma concentrations of tumour markers in the absence of neoplasia. Clinical Chemistry and Laboratory Medicine 49:1605–1620.
