• Cavernous hemangioma
• Endometrial
• Polyp
• Gynecopathology

Dear editor:

Cavernous hemangioma is an extremely rare disease in female genital tract, most of which diffusely involves the myometrium. Localized cavernous hemangioma of the uterus is even more uncommon. We hereby report a new bona file case of endometrial cavernous hemangiomatous polyp in a 34-yr-old woman. The diagnosis of this patient was confirmed by histopathologic examination. This case is of interest because it indicates that this condition could be found in the vagina, mimicking cervical polyp. Based on previous research articles,^1,2 this is the third report of endometrial cavernous hemangiomatous polyp, and it is also the first reported one that presenting as a cervical polyp. Clinicians should keep in mind that a cervical lesion may not only originate from the cervix, rather, the differential diagnosis should be broadened to include cavernous hemangioma, especially when bleeding occurs.

The patient is a 34-yr-old woman, G3P2. She was presented with a 10-day history of small amounts of vaginal bleeding, without vaginal stabbing pain or other abnormal feelings. Her past and family histories were unremarkable. There was no history of hormone usage. Laboratory tests were within normal limits. Ultrasonography did not reveal any uterine and adnexal abnormalities. Per speculum examination of vagina revealed the 8 mm ​× ​10 mm ​× ​15mm exophytic, dark red, nonfriable, polypoid mass blocked the opening of the cervix. A provisional diagnosis of cervical polyp was made. The mass was manually removed easily. It was most likely due to torsion of the pedicle. Subsequently, hysteroscopic examination showed a 3 mm stump of the stalk located in the lower uterine segment, at the 10 o'clock position of the cervical internal os. It was removed as well. Clinically, the mass was diagnosed as a cervical polyp. The patient recovered uneventfully after the polyp was removed.

The polypoid mass and the stump were submitted to Pathology Department of the Beijing Wuzhou Women and Children's Hospital for analysis. Grossly, the mass was found to be a polypoid well-circumscribed dark-red mass, 8mm ​× ​10 mm ​× ​15 mm in size. Cross section showed spongy red tissue.

Microscopically, the whole mount view of slide revealed numerous thin-walled vessels in variable sizes in a background of proliferative endometrium (Fig. 1). The vascular channels were lined by benign flat endothelial cells. The proliferative endometrial glands and stroma were scattered between the vessels (Fig. 2A). The superficial epithelium fell off partially, containing a few inflammatory cells (Fig. 2B). The endothelial nature of the lining was confirmed by the immunohistochemistry, which showed strong immunoreactivity for CD34 (Fig. 2C) and CD31 antigens. The muscle layer of the vessel wall expressed smooth muscle actin (SMA) (Fig. 2D) and desmin positivity. Other immunostaining results included CD10 positive for the endometrial stromal cells. Endothelial cells were negative for WT1 and estrogen receptor as well as progesterone receptor. The final diagnosis made was endometrial cavernous hemangiomatous polyp.

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Fig. 2

Microscopically morphology and immunohistochemical staining: endometrial tissue was seen between the vessels (Fig. 2A); part of the covering epithelium fell out (Fig. 2B); CD34 expression (Fig. 2C); SMA expression (Fig. 2D).

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Fig. 1

Cursory glance of the whole mount view of the polyp (top) and stump (bottom).

Hemangioma is a ubiquitous benign vascular tumor that seldomly involves the female genital tract, and rarely appears in the uterus. Its exact incidence is still unclear because of the very low number of cases reported. A survey of the current literature identified fewer than 50 cases of hemangioma of the uterus, and only 9 cases of cavernous hemangioma on the pregnant uterus have been published,^3,4 with the first case reported in the autopsy 1897. One of the largest series reports was constructed by Karpathiou et al.⁵ which reported the findings of nineteen vascular lesions in the gynecological tract among 50,000 gynecological tract specimens through a 20-year period, including six ovarian, two tubal, five vulvar/vaginal and six cervical lesions, whereas no lesions of the uterine corpus were detected. We searched the PubMed database and retrieved more than 50 cases of uterine and cervical cavernous hemangioma reported thus far, and most of them were case reports.

Hemangiomas are classified into two types, the congenital and acquired types. Most hemangiomas are thought to be congenital arising from the embryonic sequestration of the mesodermal tissue, rare cases of uterine hemangiomas are associated with some hereditary syndromes. Hemangiomas come in two forms: capillary and cavernous. Both forms, capillary and cavernous, are found in the uterus. Capillary hemangiomas are usually restricted to the endometrium, whereas the cavernous form affects all coats of the uterus in a diffuse fashion. The capillary form is demonstrated by small sized capillary vessels, and it is usually restricted to the endometrium,⁶ whereas the cavernous form is composed of large dilated vascular channels and involves the uterus in a diffuse fashion.⁷ Cavernous hemangiomas may be diffuse,^4,7 or localized.^6,8 The difference between the two forms is not clear, and a transitional stage may exist. It is unclear whether this lesion begins as localized and later becomes diffuse through a particular molecular pathway.

Endometrial hemangiomas are extremely rare, possibly because of the periodic endometrial shedding. Theoretically, this vascular lesion may originate from angiomatous proliferation in a polypoid endometrial lesion that persists for a prolonged period.

The diffuse form usually involves all layers of the uterus, whereas a localized hemangioma can rarely present as an endometrial polyp.^1,2 Though rare, it may be an important differential diagnosis in any female patient who presents with non-responsive uterine bleeding and/or unremitting pelvic pain.

A cavernous hemangioma localized to a portion of the uterus may be clinically silent thus making it difficult to detect. The lesion in most patients are small and asymptomatic, which could be only diagnosed by incidental findings, but sometimes it may cause abnormal vaginal bleeding or even life-threatening bleeding. It carries significant risk to the mother as well as the baby due to excessive bleeding.^9,10 Lesions are also associated with numerous obstetric and gynecological complications, but in gynecological practice, these patients mainly present with recurrent menorrhagia. Hence, it should be included in differential diagnosis of patients with vaginal bleeding.

The patient was a 34-yr-old woman, and the polyp was located in the lower uterine segment and dashed into the vagina. For the current case, the polyp was an incidental finding at speculum examination for small amounts of vaginal bleeding in a 34-yr-old woman. The epithelium covering the cavernous hemangioma having a focally erosion, which creates the lesion that bleeds when slightly contacted or damaged. Our case showed that the endometrial cavernous hemangiomatous polyp prolapsed into the introitus, mimicking a cervical polyp. The present case highlights an additional presentation of vaginal extension. This case is of interest because it indicates that this condition can charge into vagina via cervical canals mimicking a cervical polyp.

According to the last changes in terminology approved by the International Society for the Study of Vascular Anomalies (ISSVA) classification, “cavernous hemangiomas” are now called venous malformations, but anomalies of ISSVA classification is not widely used in gynecologic pathology. As a result, we still use the old term of cavernous hemangioma here.

In conclusion, endometrial cavernous hemangiomatous polyp is an extremely rare condition. Additionally, the polyp may protrude into vagina, mimicking a cervical polyp, like in our case. We present our case to the reported cases in the literature. Although this type of tumor is rare, it should be considered in the differential diagnosis of the cervical lesions when encountered in clinical practice.