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Color Doppler ultrasound in ovarian fibrosarcoma
Gynecologic Oncology 94 (2004) 229 – 231 www.elsevier.com/locate/ygyno
Case Report
Color Doppler ultrasound in ovarian fibrosarcoma Hakan Kaya, a,* Mekin Sezik, a Okan Ozkaya, a Raziye Desdicioglu, a and Nilgun Kapucuoglu b a
Department of Obstetrics and Gynecology, Suleyman Demirel University, School of Medicine, Isparta, Turkey b Department of Pathology, Suleyman Demirel University, School of Medicine, Isparta, Turkey Received in revised form 9 January 2004 Available online 18 May 2004
Abstract Background. Primary ovarian fibrosarcoma is a very rare tumor. Its Doppler waveform characteristics have not been described before. Case. A 35-year-old woman presented with a 5-cm solid ovarian mass. Intratumoral artery resistance index (RI) and pulsatility index (PI) were very low (0.19 and 0.21, respectively). Peak systolic velocity calculated by using transvaginal Doppler ultrasound was higher than expected (24.8 cm/s). Postoperatively, the histopathologic diagnosis was primary ovarian fibrosarcoma, stage Ia. Conclusions. Low vascular resistance can be encountered in ovarian fibrosarcomas. In young patients presenting with a solid adnexal mass, intratumoral Doppler waveform investigations might offer some help for earlier prediction of rare malignant tumors like fibrosarcomas. D 2004 Elsevier Inc. All rights reserved. Keywords: Fibrosarcomas; Doppler waveform; Ultrasound
Introduction Primary ovarian sarcomas represent a heterogeneous group comprising <3% of all ovarian tumors [1]. Fibrosarcoma, on the other hand, is an unusual type of ovarian sarcomas and is an exceedingly rare tumor [2]. Doppler waveform analysis of ovarian tumor blood flow by transvaginal ultrasonography may help to differentiate malignant from benign tumors of the ovary. Currently in the English literature, there is no report of an ovarian fibrosarcoma with its intratumoral artery Doppler waveform analysis available. In our report, we describe a young patient presenting with very low intratumoral resistance (RI) and pulsatility (PI) index who was later diagnosed as ovarian fibrosarcoma. Case A 35-year-old white female was initially evaluated for lower quadrant abdominal pain. Her family history is insignificant. Physical and pelvic examination revealed a left * Corresponding author. Suleyman Demirel Universitesi Tip Fakultesi, Kadin Hastaliklari ve Dogum Anabilim Dali, 32000 Isparta, Turkey. Fax: +90-246-237-1762. E-mail address: [email protected] (H. Kaya). 0090-8258/$ - see front matter D 2004 Elsevier Inc. All rights reserved. doi:10.1016/j.ygyno.2004.04.010
adnexal mass with firm and regular surface. Abdominopelvic computerized tomography demonstrated a 50 55 50 mm in diameter, solid and regular mass in the left ovarian region. Ascites was not present, and the pelvic lymph nodes were negative. Transvaginal ultrasound examination performed on cycle day 8 showed an echo-complex mass on left adnexa. The intratumoral artery RI and PI measured by transvaginal Doppler investigations were 0.19 and 0.21, respectively. Peak systolic velocity was measured as 24.8 cm/s (Fig. 1). Tumor markers including CA 125, CA 19-9, and CEA were within normal limits. The patient underwent laparoscopic left oophorectomy. Frozen section result reported an ovarian fibrosarcoma. Accordingly, laparotomy with total abdominal hysterectomy, right salpingo-oopherectomy, left salpingectomy, total omentectomy, and bilateral pelvic lymph node dissection was carried out. The final pathology report described a 55 mm in diameter, yellow-brownish, solid, firm mass confined to its capsule. Microscopically parallel intersecting spindle cell bundles, which were well demarcated from the ovarian parenchyma, were observed (Fig. 2). The spindle cells demonstrated moderate pleomorphism (Fig. 3) and increased mitotic activity (five to seven mitoses per 10 high-power fields). Necrosis was not observed. To demonstrate the mesenchymal origin of the tumor, immunostaining
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H. Kaya et al. / Gynecologic Oncology 94 (2004) 229–231
Fig. 1. Transvaginal color Doppler imaging of the solid ovarian tumor showing low-resistance and high-velocity flow (RI = 0.19, PI = 0.21) in an intratumoral vessel.
with vimentin was performed. Diffuse cytoplasmic staining was detected with vimentin in spindle cells. Nuclear staining for estrogen and progesterone receptors was negative. Proliferative index by using Ki-67 staining (Fig. 4) was 10.7%, supporting the diagnosis of fibrosarcoma. Omentum and pelvic lymph nodes were negative for metastasis. The disease was labeled as stage Ia fibrosarcoma of the ovary. In view of the patient’s stage, she was not treated with adjuvant chemotherapy. Six months postoperatively, the patient is alive with no recurrences.
Discussion
entiation from cellular fibroma may be difficult clinically and histologically [2]. Ki-67 and PCNA immunostaining assess the proliferative activity and can be used to differentiate ovarian fibrosarcomas from cellular fibromas. A proliferative index (number of cells in S + G2 + M phase) above a ratio of 6.5– 7.5 was found to correspond to malignant fibrosarcomas [3]. In our case, increased mitotic activity (>5 mitoses per 10 high-power fields) and proliferative index (10.7%) supported the diagnosis of fibrosarcoma. Although ovarian fibrosarcomas usually occur in older women, an 8-year-old girl with stage II disease was reported [4]. Our patient’s age was 35. In the literature, we were able to identify only two patients (aged 17 and
Primary ovarian fibrosarcoma is an exceedingly rare tumor arising from the basic gonadal stroma [1]. Differ-
Fig. 2. Fibrosarcoma with streaming, parallel long bundles of spindle cells (100, H&E stain).
Fig. 3. Fibrosarcoma displaying mitotic figures and moderate pleomorphism (400, H&E stain).
H. Kaya et al. / Gynecologic Oncology 94 (2004) 229–231
Fig. 4. Positive immunohistochemical staining with Ki-67 (400). The proliferative index is 10.7%.
231
nign ascites presenting with transvaginal color Doppler sonography tumor vasculature, suggesting malignancy [13]. Hence, distinguishing a cellular fibroma from fibrosarcoma by using Doppler investigations may not always be possible. Since primary ovarian fibrosarcoma is very rare, information regarding its sonomorphological characteristics is lacking. As a result, intraovarian RI, PI, and peak systolic flow values determined by color Doppler studies might be useful to detect rare ovarian malignancies like fibrosarcomas at an earlier stage, especially in younger patients presenting with a solid adnexal mass. However, the present single case would not allow us to draw further conclusions. References
35 years) with a diagnosis of ovarian fibrosarcoma <40 years of age [1,5]. Fibrosarcomas are often large tumors. The size of tumor might be helpful to differentiate fibrosarcoma from cellular fibroma [2]. However, smaller fibrosarcomas have been described [1]. One tumor, which was found on routine pelvic examination, was reported to measure only 3 cm in greatest dimension [1]. Our patient’s younger age and relatively small tumor size were also unusual for an ovarian fibrosarcoma. The intratumoral artery RI represents the blood flow impedance distal to the sampling point. Malignant tumors of the ovary have significantly lower vascular resistance than benign tumors [6]. This holds true for both PI as well as RI measurements. PI < 1 was reported to have 67% sensitivity and 97% specificity for predicting malignant ovarian tumors [7]. Peak systolic velocity values in malignant tumors are usually high, being between 20 and 60 cm/s [8]. This low-impedance, high-velocity flow is secondary to tumoral neovascularization with increased capillary permeability and altered basement membrane structure [9,10]. Increased permeability is the result of the formation of a network of capillaries whose walls are devoid of smooth muscle cells and elastic fibers. RI values have also been reported to show a strong positive correlation with the fraction of arterioles that determine the vascular resistance [6]. Fibrosarcomas are hypercellular and highly vascular tumors with evidence of intratumoral hemorrhage [11]. To our knowledge, color Doppler investigations of an ovarian fibrosarcoma have not been reported before. In their series, Tekay and Jouppila [8] reported a sarcomatic granulosa cell tumor with an RI of 0.5 and PI of 0.7. In a study with 45 patients who have complex adnexal lesions, Kupesic and Kurjak [12] misdiagnosed an ovarian fibroma as a malignant tumor using contrast-enhanced power Doppler sonography. Another case report described a postmenopausal woman with luteinized ovarian thecoma and be-
[1] Shakfeh SM, Woodruff JD. Primary ovarian sarcomas: report of 46 cases and review of the literature. Obstet Gynecol Surv 1987; 42:331 – 49. [2] Huang YC, Hsu KF, Chou CY, Dai YC, Tzeng CC. Ovarian fibrosarcoma with long-term survival: a case report. Int J Gynecol Cancer 2001;11:331 – 3. [3] Tsuji T, Kawauchi S, Utsunomiya T, Nagata Y, Tsuneyoshi M. Fibrosarcoma versus cellular fibroma of the ovary: a comparative study of their proliferative activity and chromosome aberrations using MIB-1 immunostaining, DNA flow cytometry, and fluorescence in situ hybridization. Am J Surg Pathol 1997;21:52 – 9. [4] Kraemer BB, Silva EG, Sneige N. Fibrosarcoma of ovary. A new component in the nevoid basal-cell carcinoma syndrome. Am J Surg Pathol 1984;8:231 – 6. [5] Kiyozuka Y, Nishimura H, Iwanaga S, Yakushiji M, Ito K, Nakano S, et al. Establishment and characterization of human ovarian fibrosarcoma cell line and its sensitivity to anticancer agents. [Abstract] Nippon Sanka Fujinka Gakkai Zasshi 1992;44:461 – 8. [6] Kidron D, Bernheim J, Aviram R, Cohen I, Fishman A, Beyth Y, et al. Resistance to blood flow in ovarian tumors: correlation between resistance index and histological pattern of vascularization. Ultrasound Obstet Gynecol 1999;13:425 – 30. [7] Buckshee K, Temsu I, Bhatla N, Deka D. Pelvic examination, transvaginal ultrasound and transvaginal color Doppler sonography as predictors of ovarian cancer. Int J Gynaecol Obstet 1998; 61:51 – 7. [8] Tekay A, Jouppila P. Controversies in assessment of ovarian tumors with transvaginal color Doppler ultrasound. Acta Obstet Gynecol Scand 1996;75:316 – 29. [9] Kurjak A, Kupesic S, Simunic V. Ultrasonic assessment of the periand postmenopausal ovary. Maturitas 2002;41:245 – 54. [10] Kurjak A, Jukic S, Kupesic S, Dabic D. A combined Doppler and morphopathological study of ovarian tumors. Eur J Obstet Gynecol Reprod Biol 1997;71:147 – 50. [11] Celyk C, Gungor S, Gorkemly H, Bala A, Capar M, Colakodlu M, et al. Ovarian fibrosarcomas. Acta Obstet Gynecol Scand 2002; 81:375 – 6. [12] Kupesic S, Kurjak A. Contrast-enhanced, three-dimensional power Doppler sonography for differentiation of adnexal masses. Obstet Gynecol 2000;96:452 – 8. [13] Williams LL, Fleischer AC, Jones HW. Transvaginal color Doppler sonography and CA-125 elevation in a patient with ovarian thecoma and ascites. Gynecol Oncol 1992;46:115 – 8.
Case Report
Color Doppler ultrasound in ovarian fibrosarcoma Hakan Kaya, a,* Mekin Sezik, a Okan Ozkaya, a Raziye Desdicioglu, a and Nilgun Kapucuoglu b a
Department of Obstetrics and Gynecology, Suleyman Demirel University, School of Medicine, Isparta, Turkey b Department of Pathology, Suleyman Demirel University, School of Medicine, Isparta, Turkey Received in revised form 9 January 2004 Available online 18 May 2004
Abstract Background. Primary ovarian fibrosarcoma is a very rare tumor. Its Doppler waveform characteristics have not been described before. Case. A 35-year-old woman presented with a 5-cm solid ovarian mass. Intratumoral artery resistance index (RI) and pulsatility index (PI) were very low (0.19 and 0.21, respectively). Peak systolic velocity calculated by using transvaginal Doppler ultrasound was higher than expected (24.8 cm/s). Postoperatively, the histopathologic diagnosis was primary ovarian fibrosarcoma, stage Ia. Conclusions. Low vascular resistance can be encountered in ovarian fibrosarcomas. In young patients presenting with a solid adnexal mass, intratumoral Doppler waveform investigations might offer some help for earlier prediction of rare malignant tumors like fibrosarcomas. D 2004 Elsevier Inc. All rights reserved. Keywords: Fibrosarcomas; Doppler waveform; Ultrasound
Introduction Primary ovarian sarcomas represent a heterogeneous group comprising <3% of all ovarian tumors [1]. Fibrosarcoma, on the other hand, is an unusual type of ovarian sarcomas and is an exceedingly rare tumor [2]. Doppler waveform analysis of ovarian tumor blood flow by transvaginal ultrasonography may help to differentiate malignant from benign tumors of the ovary. Currently in the English literature, there is no report of an ovarian fibrosarcoma with its intratumoral artery Doppler waveform analysis available. In our report, we describe a young patient presenting with very low intratumoral resistance (RI) and pulsatility (PI) index who was later diagnosed as ovarian fibrosarcoma. Case A 35-year-old white female was initially evaluated for lower quadrant abdominal pain. Her family history is insignificant. Physical and pelvic examination revealed a left * Corresponding author. Suleyman Demirel Universitesi Tip Fakultesi, Kadin Hastaliklari ve Dogum Anabilim Dali, 32000 Isparta, Turkey. Fax: +90-246-237-1762. E-mail address: [email protected] (H. Kaya). 0090-8258/$ - see front matter D 2004 Elsevier Inc. All rights reserved. doi:10.1016/j.ygyno.2004.04.010
adnexal mass with firm and regular surface. Abdominopelvic computerized tomography demonstrated a 50 55 50 mm in diameter, solid and regular mass in the left ovarian region. Ascites was not present, and the pelvic lymph nodes were negative. Transvaginal ultrasound examination performed on cycle day 8 showed an echo-complex mass on left adnexa. The intratumoral artery RI and PI measured by transvaginal Doppler investigations were 0.19 and 0.21, respectively. Peak systolic velocity was measured as 24.8 cm/s (Fig. 1). Tumor markers including CA 125, CA 19-9, and CEA were within normal limits. The patient underwent laparoscopic left oophorectomy. Frozen section result reported an ovarian fibrosarcoma. Accordingly, laparotomy with total abdominal hysterectomy, right salpingo-oopherectomy, left salpingectomy, total omentectomy, and bilateral pelvic lymph node dissection was carried out. The final pathology report described a 55 mm in diameter, yellow-brownish, solid, firm mass confined to its capsule. Microscopically parallel intersecting spindle cell bundles, which were well demarcated from the ovarian parenchyma, were observed (Fig. 2). The spindle cells demonstrated moderate pleomorphism (Fig. 3) and increased mitotic activity (five to seven mitoses per 10 high-power fields). Necrosis was not observed. To demonstrate the mesenchymal origin of the tumor, immunostaining
230
H. Kaya et al. / Gynecologic Oncology 94 (2004) 229–231
Fig. 1. Transvaginal color Doppler imaging of the solid ovarian tumor showing low-resistance and high-velocity flow (RI = 0.19, PI = 0.21) in an intratumoral vessel.
with vimentin was performed. Diffuse cytoplasmic staining was detected with vimentin in spindle cells. Nuclear staining for estrogen and progesterone receptors was negative. Proliferative index by using Ki-67 staining (Fig. 4) was 10.7%, supporting the diagnosis of fibrosarcoma. Omentum and pelvic lymph nodes were negative for metastasis. The disease was labeled as stage Ia fibrosarcoma of the ovary. In view of the patient’s stage, she was not treated with adjuvant chemotherapy. Six months postoperatively, the patient is alive with no recurrences.
Discussion
entiation from cellular fibroma may be difficult clinically and histologically [2]. Ki-67 and PCNA immunostaining assess the proliferative activity and can be used to differentiate ovarian fibrosarcomas from cellular fibromas. A proliferative index (number of cells in S + G2 + M phase) above a ratio of 6.5– 7.5 was found to correspond to malignant fibrosarcomas [3]. In our case, increased mitotic activity (>5 mitoses per 10 high-power fields) and proliferative index (10.7%) supported the diagnosis of fibrosarcoma. Although ovarian fibrosarcomas usually occur in older women, an 8-year-old girl with stage II disease was reported [4]. Our patient’s age was 35. In the literature, we were able to identify only two patients (aged 17 and
Primary ovarian fibrosarcoma is an exceedingly rare tumor arising from the basic gonadal stroma [1]. Differ-
Fig. 2. Fibrosarcoma with streaming, parallel long bundles of spindle cells (100, H&E stain).
Fig. 3. Fibrosarcoma displaying mitotic figures and moderate pleomorphism (400, H&E stain).
H. Kaya et al. / Gynecologic Oncology 94 (2004) 229–231
Fig. 4. Positive immunohistochemical staining with Ki-67 (400). The proliferative index is 10.7%.
231
nign ascites presenting with transvaginal color Doppler sonography tumor vasculature, suggesting malignancy [13]. Hence, distinguishing a cellular fibroma from fibrosarcoma by using Doppler investigations may not always be possible. Since primary ovarian fibrosarcoma is very rare, information regarding its sonomorphological characteristics is lacking. As a result, intraovarian RI, PI, and peak systolic flow values determined by color Doppler studies might be useful to detect rare ovarian malignancies like fibrosarcomas at an earlier stage, especially in younger patients presenting with a solid adnexal mass. However, the present single case would not allow us to draw further conclusions. References
35 years) with a diagnosis of ovarian fibrosarcoma <40 years of age [1,5]. Fibrosarcomas are often large tumors. The size of tumor might be helpful to differentiate fibrosarcoma from cellular fibroma [2]. However, smaller fibrosarcomas have been described [1]. One tumor, which was found on routine pelvic examination, was reported to measure only 3 cm in greatest dimension [1]. Our patient’s younger age and relatively small tumor size were also unusual for an ovarian fibrosarcoma. The intratumoral artery RI represents the blood flow impedance distal to the sampling point. Malignant tumors of the ovary have significantly lower vascular resistance than benign tumors [6]. This holds true for both PI as well as RI measurements. PI < 1 was reported to have 67% sensitivity and 97% specificity for predicting malignant ovarian tumors [7]. Peak systolic velocity values in malignant tumors are usually high, being between 20 and 60 cm/s [8]. This low-impedance, high-velocity flow is secondary to tumoral neovascularization with increased capillary permeability and altered basement membrane structure [9,10]. Increased permeability is the result of the formation of a network of capillaries whose walls are devoid of smooth muscle cells and elastic fibers. RI values have also been reported to show a strong positive correlation with the fraction of arterioles that determine the vascular resistance [6]. Fibrosarcomas are hypercellular and highly vascular tumors with evidence of intratumoral hemorrhage [11]. To our knowledge, color Doppler investigations of an ovarian fibrosarcoma have not been reported before. In their series, Tekay and Jouppila [8] reported a sarcomatic granulosa cell tumor with an RI of 0.5 and PI of 0.7. In a study with 45 patients who have complex adnexal lesions, Kupesic and Kurjak [12] misdiagnosed an ovarian fibroma as a malignant tumor using contrast-enhanced power Doppler sonography. Another case report described a postmenopausal woman with luteinized ovarian thecoma and be-
[1] Shakfeh SM, Woodruff JD. Primary ovarian sarcomas: report of 46 cases and review of the literature. Obstet Gynecol Surv 1987; 42:331 – 49. [2] Huang YC, Hsu KF, Chou CY, Dai YC, Tzeng CC. Ovarian fibrosarcoma with long-term survival: a case report. Int J Gynecol Cancer 2001;11:331 – 3. [3] Tsuji T, Kawauchi S, Utsunomiya T, Nagata Y, Tsuneyoshi M. Fibrosarcoma versus cellular fibroma of the ovary: a comparative study of their proliferative activity and chromosome aberrations using MIB-1 immunostaining, DNA flow cytometry, and fluorescence in situ hybridization. Am J Surg Pathol 1997;21:52 – 9. [4] Kraemer BB, Silva EG, Sneige N. Fibrosarcoma of ovary. A new component in the nevoid basal-cell carcinoma syndrome. Am J Surg Pathol 1984;8:231 – 6. [5] Kiyozuka Y, Nishimura H, Iwanaga S, Yakushiji M, Ito K, Nakano S, et al. Establishment and characterization of human ovarian fibrosarcoma cell line and its sensitivity to anticancer agents. [Abstract] Nippon Sanka Fujinka Gakkai Zasshi 1992;44:461 – 8. [6] Kidron D, Bernheim J, Aviram R, Cohen I, Fishman A, Beyth Y, et al. Resistance to blood flow in ovarian tumors: correlation between resistance index and histological pattern of vascularization. Ultrasound Obstet Gynecol 1999;13:425 – 30. [7] Buckshee K, Temsu I, Bhatla N, Deka D. Pelvic examination, transvaginal ultrasound and transvaginal color Doppler sonography as predictors of ovarian cancer. Int J Gynaecol Obstet 1998; 61:51 – 7. [8] Tekay A, Jouppila P. Controversies in assessment of ovarian tumors with transvaginal color Doppler ultrasound. Acta Obstet Gynecol Scand 1996;75:316 – 29. [9] Kurjak A, Kupesic S, Simunic V. Ultrasonic assessment of the periand postmenopausal ovary. Maturitas 2002;41:245 – 54. [10] Kurjak A, Jukic S, Kupesic S, Dabic D. A combined Doppler and morphopathological study of ovarian tumors. Eur J Obstet Gynecol Reprod Biol 1997;71:147 – 50. [11] Celyk C, Gungor S, Gorkemly H, Bala A, Capar M, Colakodlu M, et al. Ovarian fibrosarcomas. Acta Obstet Gynecol Scand 2002; 81:375 – 6. [12] Kupesic S, Kurjak A. Contrast-enhanced, three-dimensional power Doppler sonography for differentiation of adnexal masses. Obstet Gynecol 2000;96:452 – 8. [13] Williams LL, Fleischer AC, Jones HW. Transvaginal color Doppler sonography and CA-125 elevation in a patient with ovarian thecoma and ascites. Gynecol Oncol 1992;46:115 – 8.