Journal of Obstetrics and Gynaecology
ISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20
Ovarian haemangioma with hyperandrogenism: a case report K. Shitsukawa, S. Shinomiya, Y. Tanaka, M. Okada & M. Kamada To cite this article: K. Shitsukawa, S. Shinomiya, Y. Tanaka, M. Okada & M. Kamada (2015) Ovarian haemangioma with hyperandrogenism: a case report, Journal of Obstetrics and Gynaecology, 35:2, 214-215, DOI: 10.3109/01443615.2014.940288 To link to this article: http://dx.doi.org/10.3109/01443615.2014.940288
Published online: 24 Jul 2014.
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Figure 1. Partial vulvectomy procedure. (A, B) The vulva prior to and following surgery with the intradermal nevi resected, respectively. (C–E) Surgical procedure and a Foley catheter that was inserted to assure urethral patency. (F, G) Excised specimen with a basal incision to show the metastasis.
Julien V, Labadie M, Gauthier G et al. 2012. Clitoral metastasis from ductal breast cancer revealing metastases in multiple sites and review of the literature. Journal of Lower Genital Tract Disease 16:66–69. Kobayashi F, Tanada S, Mizuno Y et al. 1999. A case of clitoral metastasis without skin involvement from rectal cancer. Gynecologic Oncology 74:138–140. Langenstroer P, Romanas M, Damjanov I. 2003. Clitoromegaly caused by metastatic carcinosarcoma of the urinary bladder. Archives of Pathology and Laboratory Medicine 127:505. Matsuo K, Hew KE, Im, DD et al. 2009. Clitoral metastasis of anal adenocarcinoma associated with rectovaginal fistula in long standing Crohn’s disease. European Journal of Obstetrics, Gynecology, and Reproductive Biology 144:182–183. Rampal M, Hermanowicz M. 1973. [Clitoral metastases of a kidney cancer]. Journal D’urologie et de Nephrologie 79:386. Thomas JA, Matanhelia SS, Griffiths D et al. 1994. Clitoral metastasis secondary to transitional cell carcinoma of the bladder. British Journal of Urology 74:524–525. Yazgan C, Erden A, Yagci C et al. 2004. Clitoral metastasis from transitional cell carcinoma of the renal pelvis: CT and MRI findings. International Urology and Nephrology 36:331–333.
Ovarian haemangioma with hyperandrogenism: a case report K. Shitsukawa1, S. Shinomiya2, Y. Tanaka1, M. Okada1 & M. Kamada1 Departments of 1Obstetrics and Gynecology and 2Pathology, Tokushima Prefecture Naruto Hospital, Naruto, Tokushima, Japan DOI: 10.3109/01443615.2014.940288 Correspondence: K. Shitsukawa, Department of Obstetrics and Gynecology, Tokushima Prefecture Naruto Hospital, 32 Aza-Kotani, Kurosaki Muya-cho, Naruto, Tokushima 772-8503, Japan. E-mail: [email protected]
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).
Introduction
Downloaded by [Universite Laval] at 12:37 15 September 2015
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.
ISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20
Ovarian haemangioma with hyperandrogenism: a case report K. Shitsukawa, S. Shinomiya, Y. Tanaka, M. Okada & M. Kamada To cite this article: K. Shitsukawa, S. Shinomiya, Y. Tanaka, M. Okada & M. Kamada (2015) Ovarian haemangioma with hyperandrogenism: a case report, Journal of Obstetrics and Gynaecology, 35:2, 214-215, DOI: 10.3109/01443615.2014.940288 To link to this article: http://dx.doi.org/10.3109/01443615.2014.940288
Published online: 24 Jul 2014.
Submit your article to this journal
Article views: 23
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: [Universite Laval]
Date: 15 September 2015, At: 12:37
Gynaecology Case Reports
Downloaded by [Universite Laval] at 12:37 15 September 2015
214
Figure 1. Partial vulvectomy procedure. (A, B) The vulva prior to and following surgery with the intradermal nevi resected, respectively. (C–E) Surgical procedure and a Foley catheter that was inserted to assure urethral patency. (F, G) Excised specimen with a basal incision to show the metastasis.
Julien V, Labadie M, Gauthier G et al. 2012. Clitoral metastasis from ductal breast cancer revealing metastases in multiple sites and review of the literature. Journal of Lower Genital Tract Disease 16:66–69. Kobayashi F, Tanada S, Mizuno Y et al. 1999. A case of clitoral metastasis without skin involvement from rectal cancer. Gynecologic Oncology 74:138–140. Langenstroer P, Romanas M, Damjanov I. 2003. Clitoromegaly caused by metastatic carcinosarcoma of the urinary bladder. Archives of Pathology and Laboratory Medicine 127:505. Matsuo K, Hew KE, Im, DD et al. 2009. Clitoral metastasis of anal adenocarcinoma associated with rectovaginal fistula in long standing Crohn’s disease. European Journal of Obstetrics, Gynecology, and Reproductive Biology 144:182–183. Rampal M, Hermanowicz M. 1973. [Clitoral metastases of a kidney cancer]. Journal D’urologie et de Nephrologie 79:386. Thomas JA, Matanhelia SS, Griffiths D et al. 1994. Clitoral metastasis secondary to transitional cell carcinoma of the bladder. British Journal of Urology 74:524–525. Yazgan C, Erden A, Yagci C et al. 2004. Clitoral metastasis from transitional cell carcinoma of the renal pelvis: CT and MRI findings. International Urology and Nephrology 36:331–333.
Ovarian haemangioma with hyperandrogenism: a case report K. Shitsukawa1, S. Shinomiya2, Y. Tanaka1, M. Okada1 & M. Kamada1 Departments of 1Obstetrics and Gynecology and 2Pathology, Tokushima Prefecture Naruto Hospital, Naruto, Tokushima, Japan DOI: 10.3109/01443615.2014.940288 Correspondence: K. Shitsukawa, Department of Obstetrics and Gynecology, Tokushima Prefecture Naruto Hospital, 32 Aza-Kotani, Kurosaki Muya-cho, Naruto, Tokushima 772-8503, Japan. E-mail: [email protected]
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).
Introduction
Downloaded by [Universite Laval] at 12:37 15 September 2015
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.
