|Year : 2015 | Volume
| Issue : 2 | Page : 227-230
An incidental ovarian mass: A case of ovarian hemangioma with prominent stromal luteinization
Babak Shirazi1, Mohammad Hossein Anbardar1, Negar Azarpira2, Minoo Robati3
1 Department of Pathology, Shiraz University of Medical Sciences, Shiraz, Iran
2 Transplant Research Center, Shiraz University of Medical Sciences, Shiraz, Iran
3 Department of Gynecology and Obstetrics, Shiraz University of Medical Sciences, Shiraz, Iran
|Date of Web Publication||13-Mar-2015|
Mohammad Hossein Anbardar
Department of Pathology, Shiraz University of Medical Sciences, P.O. Box: 71345-1864, Shiraz
Source of Support: None, Conflict of Interest: None
Ovarian hemangioma is a rare benign tumor of female genital tract. Stromal luteinization in ovarian hemangioma is an uncommon process and the pathogenesis is controversial. In this regard, two hypotheses have been suggested whether luteinization is a reactive process or it is the stimulator for development of ovarian hemangioma. Here, we report a case of a 55-year-old woman who referred to our center due to incidental finding of left ovarian mass in pelvic sonography. Microscopically, the mass showed a mixed cavernous and capillary hemangioma and the peripheral stroma contained several small and large clusters of stromal cells, which were luteinized. It should be noted that an ovarian hemangioma could be associated with stromal luteinization although its pathogenesis is not clearly known. Yet, we believe the stromal luteinization around ovarian hemangioma could be a reactive phenomenon.
Keywords: Hemangioma, luteinization, ovary, stroma
|How to cite this article:|
Shirazi B, Anbardar MH, Azarpira N, Robati M. An incidental ovarian mass: A case of ovarian hemangioma with prominent stromal luteinization. Med J DY Patil Univ 2015;8:227-30
| Introduction|| |
Ovarian hemangioma is a rare benign tumor of female genital tract. Most ovarian hemangiomas are asymptomatic, of cavernous type, small, and incidental findings at operation or autopsy. ,,,,,,,,,, Ovarian stromal cell luteinization could be seen in different ovarian lesions whether the lesion is benign, malignant, primary, or secondary. , An ovarian hemangioma can be associated with stromal luteinization that has been reported in 11 cases in the English literature. ,,,,,,, However, its pathogenesis is not clearly known yet.
Here, we report a case of ovarian hemangioma with stromal hyperplasia and luteinization with estrogen receptor evaluation and discuss the proposed hypotheses in pathogenesis of stromal luteinization. Furthermore, the reported cases of ovarian hemangioma with stromal luteinization in the literature are going to be reviewed.
| Case Report|| |
A 55-year-old woman referred to our center due to incidental finding of left ovarian mass in pelvic sonography. She was para 2, and menopause without any symptoms, such as uterine bleeding, discharge, pain, and weight loss. Her past medical and family history was unremarkable. Nonetheless, a small painless mobile firm and solid left adnexal mass was detected in the bimanual pelvic examination. Pelvic sonography also showed enlarged left ovary (40 mm × 21 mm) without visible mass. In addition, pelvic magnetic resonance imaging with contrast showed an apparent round lesion measuring 41 mm × 30 mm in the left ovary, which was hypersignal at T2 and hyposignal at T1. It also showed significant enhancement with contrast. The patient's serum CA-125 and beta-human chorionic gonadotropin levels were normal. However, Pap smear More Details revealed atrophic changes. Due to suspicion to malignancy in imaging and during surgery, trans-abdominal hysterectomy and bilateral salpingo-oophorectomy were performed. Grossly, the left ovary measured 40 mm × 30 mm × 30 mm with surface nodularity. The cut section revealed a relatively well-defined creamy-gray mass measuring 25 mm × 20 mm × 20 mm with multiple hemorrhagic microcystic areas [Figure 1]. Besides, microscopic examination revealed a mixed cavernous and capillary hemangioma consisting of multiple various sized thin-walled blood vessels, filled with red blood cells. A single layer of the endothelium lined the vessels. The peripheral stroma contained several small and large clusters of stromal cells which were luteinized [Figure 2]. The right ovary, on the other hand, showed no mass or luteinization. However, atrophic changes were detected in the endometrium. Later, immunohistochemical study was carried out indicating the strong immune reactivity of the endothelial cells for CD31, CD34, and factor VIII-related antigen [Figure 3]a and b. However, they showed no reactivity for estrogen receptor. The luteinized stromal cells showed strong reactivity for inhibin. The patient is well 2 years after her surgery.
|Figure 2: The photomicrograph shows numerous thin-walled vascular channels with different sizes and configurations, lined by a single layer of flattened endothelial cells (lower right) and the surrounding stroma contains luteinized stromal cells (upper left) (H and E, ×400)|
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|Figure 3: (a) The endothelial cells show immunoreactivity for CD34 (IHC, ×400). (b) The luteinized cells show strong immunoreactivity for inhibin (IHC, ×400)|
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| Discussion|| |
Ovarian hemangioma is a very rare benign lesion of ovary. Most ovarian hemangiomas are usually asymptomatic, present as incidental findings during operation or autopsy, and have been reported in both children and adults.  Despite the rich vascularity of the ovary, the incidence of ovarian hemangioma is rare.  Microscopically, most of the tumors are of cavernous or mixed cavernous and capillary type. The vascular channels are variable in size and are lined by a flat single layer of endothelial cells. The lumen may contain thrombosis and the intervening stroma may show hyalinization, calcification, and occasionally luteinization. Most tumors are unilateral, under 1.5 cm in diameter, in the hilus or medulla of the ovary, and sharply separated from adjacent tissue, a feature distinguishing them from normal vascular channels in the ovary. , The etiology of ovarian hemangiomas is controversial. Some authors believed that it was a true primary pure ovarian mesenchymal neoplasm or a part of mature teratoma. Others stated that the hemangiomas were hamartomatous malformation but not neoplastic lesions. The formation of this lesion may be stimulated by some factors, such as hormones, pregnancy, or infection. 
Although ovarian hemangiomas are nonfunctional neoplasms, associated stromal luteinization has been reported in few cases as in the present case. To the best of our knowledge, 11 cases were reported previously in the English literature and the details have been presented in [Table 1]. In nonendocrine ovarian tumors, the stromal luteinization is most commonly accompanied by ovarian epithelial tumors and this association is uncommon in nonepithelial ovarian tumors.  The pathogenesis of stromal luteinization in ovarian hemangioma is not clearly known yet. In this respect, two important hypotheses have been suggested whether luteinization is the stimulator for development of ovarian hemangioma or it is a reactive process.
|Table 1: Reported cases of ovarian hemangioma with stromal luteinization in English literature|
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The first hypothesis recommended that hyperestrogenism resulted from stromal hyperthecosis or stromal hyperplasia was the first event, which could stimulate the development of an ovarian hemangioma. Estrogen has proved growth stimulatory influences on endothelial cells and most hemangiomas express estrogen receptor on endothelial cells. ,, This hypothesis has been less supported due to three reasons. First of all, only three out of the all the studies on this issue have performed immunohistochemistry for estrogen and progesterone receptor, two of which being negative and one being positive. ,, In our case, the estrogen receptor was negative. Considering the second reason, if stromal luteinization is the consequence of hyperestrogenism, it should be bilateral.  In most cases, as in our case, however the luteinization was unilateral [Table 1]. Finally, this is evident that hyperestrogenism is very common in the population, while ovarian hemangioma is too uncommon. 
The second hypothesis is a mechanical theory suggesting that ovarian hemangioma was the primary event that stimulated stromal luteinization by expansion of the mass. ,,, This is consistent with the fact that luteinization of stromal cells is limited to the stroma of the mass or the stroma surrounding the mass. In the cases with bilateral stromal luteinization, the changes are more prominent in the ovary containing the tumor.  In our case, the hemangioma was unilateral and stromal luteinization was limited to the surrounding stroma of the tumor. These evidences support the idea that the stromal luteinization around ovarian hemangiomas could be a reactive phenomenon.
Differential diagnoses of ovarian hemangioma include normal medullary vasculature, lymphangiomas, cystic teratomas with prominent vascular component, and angiosarcomas.  Yet, presence of well-defined masses composed of variable sized vascular channels can differentiate hemangiomas from normal medullary vasculature. To rule out teratoma, thorough sampling is mandatory for excluding the presence of other teratomatous components. , Moreover, angiosarcomas have prominent cytologic atypia, mitotic figures, necrosis, and anastomosing channels that differentiate them from hemangiomas. 
| Conclusion|| |
It should be noted that an ovarian hemangioma can be associated with stromal luteinization although its pathogenesis is not clearly known. According to negative estrogen receptor and unilateral luteinization in our case, we believe that the pathogenesis is a mechanical process that leads to formation of stromal luteinization around an ovarian hemangioma.
| Acknowledgment|| |
The researchers are grateful for Ms. A. Keivanshekouh at the Research Improvement Center of Shiraz University of Medical Sciences for improving the use of English in the manuscript.
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[Figure 1], [Figure 2], [Figure 3]