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19 April 2023: Articles  Japan

A Case of a Refractory Bleeding Giant Vaginal Wall Cavernous Hemangioma Successfully Managed with Sclerotherapy

Rare disease

Mari Fukuoka1ABCDEF, Toshiyuki Okumura1ABCDEF*, Ayato Hayashi2ABCDEF, Natsumi Takeda1ABCDEF, Akari Koizumi1BDEF, Takafumi Ujihira1BDEF, Shintaro Makino1ABCDEF

DOI: 10.12659/AJCR.939474

Am J Case Rep 2023; 24:e939474

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Abstract

BACKGROUND: Vaginal wall hemangiomas are extremely rare, benign, vascular tumors of the female genitalia. Most cases occur in childhood, but a few cases can be acquired; however, the mechanism of hemangioma formation remains unknown. Most hemangiomas involving female genital organs are small and asymptomatic. However, huge hemangiomas can cause irregular genital bleeding, infertility, and miscarriage. Surgical excision and embolization are the most common treatment options. We reveal that sclerotherapy achieved good outcomes in a patient with an intractable huge vaginal wall hemangioma.

CASE REPORT: A 71-year-old woman visited a local doctor with concerns of frequent urination. A ring pessary was inserted after a diagnosis of pelvic organ prolapse. However, symptoms did not improve, and the patient consulted another hospital. The previous physician diagnosed vaginal wall tumors and prolapse and performed a colporrhaphy. However, she was referred to our hospital with heavy intraoperative bleeding. Imaging examination revealed a huge hemangioma on the vaginal wall, which was histologically diagnosed as a cavernous hemangioma. Angiography revealed hemorrhage in the right peripheral vaginal artery. Owing to concerns regarding extensive vaginal wall necrosis caused by arterial embolization, sclerotherapy using monoethanolamine oleate was selected. Hemostasis was achieved 1 month after sclerotherapy, and postoperative imaging showed the lesion had shrunk in size. No recurrence of hemangioma was observed 19 months after surgery.

CONCLUSIONS: We report a case of a large vaginal wall intractable bleeding hemangioma. Sclerotherapy can be a suitable treatment option for large vaginal hemangiomas that are too extensive to be treated using surgery or arterial embolization.

Keywords: Hemangioma, Cavernous, Sclerotherapy, Monoethanolamine Oleate, Female, Humans, Aged, Hemangioma, Vagina, Uterine Hemorrhage

Background

Hemangiomas can occur systemically, but the most common sites are the skin of the head and the neck [1]. Hemangiomas rarely occur in female genital organs, such as the uterus, vagina, and perineum [2]. Most cases are congenital and are observed in childhood [1], but some are acquired due to trauma or infection, and the mechanism of their formation is unknown [3]. It is difficult to distinguish these lesions from varicose veins or vascular malformations macroscopically; therefore, they are diagnosed histologically [4]. The pathological findings of hemangiomas include polypoid lesions of dilated blood vessels with increased vascular endothelial cells in the dermis and deep subcutaneous tissues [4]. Most hemangiomas in female genital organs are small and asymptomatic [1]; however, depending on the extent and location of the lesion, hemangiomas can cause irregular genital bleeding as well as infertility, miscarriage, intrauterine fetal death, and delivery failure due to obstruction of the ostium vaginae [4]. Vaginal wall hemangiomas in pregnant women often occur at the site of an episiotomy performed during a previous pregnancy [3]. Some cases in which hemangiomas grew during pregnancy and required treatment, especially in the last trimester, have been reported [5]. Some cases in which vaginal delivery or episiotomy was performed without a hemangioma being noticed, resulting in heavy bleeding, have also been reported [1]. Hormonal dynamics have been identified as a possible mechanism for a growing hemangioma during pregnancy [1]. Yamashita confirmed the expression of estrogen receptors, progesterone receptors, and vascular endothelial growth factor (VEGF) in endothelial cells and perivascular cells of cervical and vaginal wall hemangiomas; moreover, the increased secretion of estrogen and progesterone during pregnancy promotes the secretion of angiogenic factors, such as VEGF [3]. Hemangiomas can induce severe bleeding even with minimal invasion [1]; therefore, assessment of lesion size using color Doppler ultrasound [6], magnetic resonance imaging (MRI) [7], or angiography [6] is recommended prior to biopsy and treatment [1]. Surgical resection and embolization are the most common treatment options, and these are selected based on the location and size of the lesion [2]. In this report, we show that sclerotherapy achieved good outcomes in a patient with an intractable huge vaginal wall hemangioma with symptoms related to irregular genital bleeding and pelvic organ prolapse.

Case Report

A 71-year-old woman, gravida 4, para 3 with 2 prior uncomplicated vaginal deliveries and 1 spontaneous abortion, with no medical history of any special note, visited her local doctor with a concern of frequent urination. A ring pessary was inserted after a diagnosis of pelvic organ prolapse. However, her symptoms did not improve, and after insertion of the pessary, the patient also had irregular genital bleeding for the first time. A Pap smear performed at that time was negative for intra-epithelial lesions and malignancy. The first physician made a diagnosis of pelvic organ prolapse and referred the patient to the second doctor for surgery. The second physician diagnosed vaginal wall tumors and prolapse at both the anterior and posterior vaginal wall (Figure 1A) and then performed a colporrhaphy. During the surgery, eruptive profuse hemorrhage was noted with puncture of a vaginal wall tumor (Figure 1B). Intraoperative blood loss was 177 mL. Her vital signs were normal throughout the entire hospitalization. On postoperative day 1, there was no obvious bleeding, but a 2 point drop in hemoglobin was noted (Figure 1C), so gauze packing was applied to the surgical site. Since there was no bleeding or decrease in hemoglobin, the gauze was removed the next day and the patient was discharged 5 days after surgery. However, she experienced continuous bleeding postoperatively and was admitted again 4 days after discharge. The doctor performed hemostasis surgery. Intraoperative blood loss was 60 mL. She was discharged 2 days after reoperation but the postoperative hemorrhage was about the second day of menstruation and needed gauze packing to control bleeding. Moreover, there was slow progression of anemia (Figure 1C), and the patient was referred to our hospital for further treatment. On initial examination, persistent bleeding was observed from the 9 o’clock position on the vaginal wall (Figure 2). Pelvic dynamic computed tomography (CT) revealed a marked contrast effect in the entire vagina (Figure 3A), and MRI showed that the entire vaginal wall seemed to be replaced by a hemangioma (Figure 3C). The pathology of vaginal wall tumors resected at the time of the previous physician’s surgery showed irregularly dilated vessels of various sizes growing in a spongy pattern and thrombus formation in the vascular cavity, leading to the diagnosis of a cavernous hemangioma. Because of the continuous bleeding, equivalent to the second day of menstruation, the gauze was packed at the surgical site even after she was admitted to our hospital. With gauze compression, bleeding passed with only adhesion. Therefore, interventional radiology (IVR) was planned to determine the most appropriate treatment method. Until the date of IVR, the gauze packing was changed repeatedly every 2 to 3 days. Angiography was performed to identify the responsible artery, which was revealed to be a point in peripheral right vaginal artery (Figure 4A, 4B). The distance to the responsible artery was significant; therefore, it could not be reached with the longest catheter, and this made embolization difficult. Moreover, embolization of the right vaginal artery might have induced extensive vaginal wall necrosis.

We decided to perform sclerotherapy, a form of local treatment, in collaboration with a plastic surgeon. An occlusion catheter was inserted into the bilateral common iliac arteries to prevent the spread of the drug by rapid blood flow, and the operation was performed under the condition that blood flow could be adjusted beforehand. An oocyte retrieval needle and a needle guide was attached to the transvaginal ultrasound (Figure 5A), and 2.0 mL of monoethanolamine oleate was injected into the bleeding site at the 9 o’clock position on the vaginal wall. Because of the possibility of bleeding from the vaginal wall at the 3 o’clock position, 2.5 mL of monoethanolamine oleate was injected in the same way (Figure 5B). After sclerotherapy, bleeding was reduced to a small amount but was not completely hemostatic. Therefore, we needed to change the packing gauze every 2 to 3 days. Hemostatic agents were not used and no blood transfusions were required. Hemostasis was achieved 1 month after surgery, and the hemangioma had shrunk significantly to the point that it could not be measured by CT at 2 months after surgery and MRI at 4 months after. There was no recurrence of hemangioma at 1 year and 7 months after surgery (Figure 3B, 3D).

Discussion

In this case report, we showed sclerotherapy can be a suitable treatment option for large vaginal hemangiomas with intractable bleeding. Surgical excision is the best option, especially for small hemangiomas [8,9]. There are some problems with this option, such as bleeding [9]. When there is concern about heavy bleeding, such as when the lesion is large or deep, embolization is often performed [10]. In the present case, the hemangioma was too large to do embolization because the embolization can induce necrosis in her whole vagina, and removal of the entire vaginal wall would have been too invasive. To the best of our knowledge, there are no reports of sclerotherapy treating hemangiomas of the female genitalia that are so large that the vaginal wall appears to be replaced by a hemangioma. There have been reports of surgical resection of giant hepatic hemangiomas after embolization to control bleeding [10]. However, in the present case, sclerotherapy was used to treat local lesions because selective embolization of the responsible artery was difficult and extensive vaginal wall necrosis might have occurred. We used monoethanolamine oleate for sclerotherapy because there is a lot of experience in its use as the treatment method for varicose veins, esophageal varix, and gastric varix and it has recently been used for venous vascular malformations [8,9]. Monoethanolamine oleate acts by damaging the vascular intima, which leads to thrombus formation [11]. It is an anionic surfactant that acts on and destroys the lipid portion of the vascular endothelial cells [12], and deposition and accumulation of fibrin, platelets, and erythrocytes occur in exposed subendothelial tissues. Thrombus organization takes about 4 weeks, and the hemangioma gradually scleroses and regresses [9]. In the present case, the bleeding stopped 1 month after sclerotherapy. Because organization and shrinkage of the lesion are achieved without necrosis or scar formation at the injection site, the patient’s cosmetic appearance is not influenced by sclerotherapy. One of the adverse effects of sclerotherapy with monoethanolamine oleate is hemoglobinuria associated with hemolysis, which occurs in approximately 30% of patients [8]. This adverse effect induces renal dysfunction, which can be treated with haptoglobin [8]. Other adverse effects include pain during injection [13], but local anesthesia was sufficient in the present case. Also, reports of anaphylactic reactions due to monoethanolamine oleate are thought to have a lesser risk (0.005% [14]) than allergic reactions to sodium morrhuate (11–48% [15]) or sodium tetradecyl sulfate (0.15% [16]).

Although several cases of vaginal wall hemangioma treated with surgery, arterial embolization, or sclerotherapy have been reported, to the best of our knowledge, this is the first report of the use of sclerotherapy in the management of a large vaginal wall hemangioma. Sclerotherapy is a local treatment and a more minimally invasive approach than surgical resections from the perspective of less blood loss, smaller surgical wound size, and shorter hospital stay [17]. In the future, sclerotherapy may be an effective treatment for vaginal wall hemangiomas that are difficult to treat using arterial embolization or surgery. However, since the number of cases of large vaginal wall hemangioma is very low, it is necessary to accumulate a larger number of cases and to investigate the safety and efficacy of this treatment.

Conclusions

Here, we report a case of a large vaginal wall hemangioma that was difficult to treat. We conclude that sclerotherapy is an effective and safe treatment option for extensive lesions, especially in cases in which conventional surgical treatment and arterial embolization are not available.

Figures

(A) Hemangioma protruding from the vagina (white arrowhead); urethral meatus (yellow arrow); labia minora (white arrow). (B) Eruptive massive hemorrhage was noted with puncture of vaginal wall hemangioma (white arrowhead). (C) Hemoglobin (Hb) change during hospitalization and visits to previous physician.Figure 1.. (A) Hemangioma protruding from the vagina (white arrowhead); urethral meatus (yellow arrow); labia minora (white arrow). (B) Eruptive massive hemorrhage was noted with puncture of vaginal wall hemangioma (white arrowhead). (C) Hemoglobin (Hb) change during hospitalization and visits to previous physician. The right vaginal wall is visible using a vaginal speculum. Bleeding from vaginal wall at 9 o’clock (white arrowhead).Figure 2.. The right vaginal wall is visible using a vaginal speculum. Bleeding from vaginal wall at 9 o’clock (white arrowhead). (A) Dynamic computed tomography scan (axial) of the pelvic region in the arterial layer showing a strong contrast effect in the entire vagina, which is suggestive of abundant blood flow (white arrowheads). Urinary bladder (white arrow), rectum (yellow arrow). (B) Two months after embolization. Vaginal wall hemangioma is obscured (white arrowhead); urinary bladder (white arrow); rectum (yellow arrow). (C) Contrast-enhanced magnetic resonance image (T2, sagittal) of the pelvic region showing the entire vaginal wall seemed to be replaced by a hemangioma with a diameter of 10 cm (white arrowheads); urinary bladder (white arrow); rectum (yellow arrow). (D) Four months after sclerotherapy; vaginal wall hemangioma became obscured; uterus (white arrow); vagina (yellow arrow).Figure 3.. (A) Dynamic computed tomography scan (axial) of the pelvic region in the arterial layer showing a strong contrast effect in the entire vagina, which is suggestive of abundant blood flow (white arrowheads). Urinary bladder (white arrow), rectum (yellow arrow). (B) Two months after embolization. Vaginal wall hemangioma is obscured (white arrowhead); urinary bladder (white arrow); rectum (yellow arrow). (C) Contrast-enhanced magnetic resonance image (T2, sagittal) of the pelvic region showing the entire vaginal wall seemed to be replaced by a hemangioma with a diameter of 10 cm (white arrowheads); urinary bladder (white arrow); rectum (yellow arrow). (D) Four months after sclerotherapy; vaginal wall hemangioma became obscured; uterus (white arrow); vagina (yellow arrow). (A) Early layer of contrast injection to the right vaginal artery angiography. (B) Leakage of contrast medium observed from the peripheral blood vessels (white arrowhead), which was considered to be the cause of the bleeding.Figure 4.. (A) Early layer of contrast injection to the right vaginal artery angiography. (B) Leakage of contrast medium observed from the peripheral blood vessels (white arrowhead), which was considered to be the cause of the bleeding. (A) Transvaginal ultrasound probe with an egg collection needle attached. The egg collection needle protrudes from the tip (white arrowheads). (B) Monoethanolamine oleate is injected and hemangioma is contrasted (white arrowhead).Figure 5.. (A) Transvaginal ultrasound probe with an egg collection needle attached. The egg collection needle protrudes from the tip (white arrowheads). (B) Monoethanolamine oleate is injected and hemangioma is contrasted (white arrowhead).

References:

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2.. Celik F, Arioz DT, Köken GN, Yilmazer M, Very rare cause of vaginal mass in pregnancy: Cavernous hemangioma: J Obstet Gynaecol Res, 2012; 38(5); 889-91

3.. Yamashita T, Takayanagi N, Higashi M, The relationship between vaginal cavernous hemangiomas and late pregnancy. A case report and a review of the literature: Clin Exp Obstet Gynecol, 2019; 46; 641-45

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5.. Chi JH, Manley GT, Chou D, Pregnancy-related vertebral hemangioma. Case report, review of the literature, and management algorithm: Neurosurg Focus, 2005; 19(3); E7

6.. Emoto M, Tamura R, Izumi H, Sonodynamic changes after transcatheter arterial embolization in a vaginal hemangioma: Case report: Ultrasound Obstet Gynecol, 1997; 10(1); 66-67

7.. Mastropasqua M, Kanematsu M, Leonardou P, Cavernous hemangiomas in patients with chronic liver disease: MR imaging findings: Magn Reson Imaging, 2004; 22(1); 15-18

8.. Fujiki M, Ozaki M, Kurachi I, Risk factors for macroscopic haemoglobinuria after sclerotherapy using ethanolamine oleate for venous malformations: Eur J Vasc Endovasc Surg, 2019; 58(1); 105-11

9.. Fernandes DT, Elias RA, Santos-Silva AR, Benign oral vascular lesions treated by sclerotherapy with ethanolamine oleate: A retrospective study of 43 patients: Med Oral Patol Oral Cir Bucal, 2018; 23(2); e180-e87

10.. Maker AV, Al Rameni D, Prabhakar N, Combining on-table embolization with immediate resection to safely excise giant hepatic hemangiomas: J Gastrointest Surg, 2021; 25(6); 1651-53

11.. Bonan PR, Miranda Lde P, Mendes DC, Effectiveness of low flow vascular lesions sclerosis with monoethanolamine: Report of six cases: Med Oral Patol Oral Cir Bucal, 2007; 12(7); E524-27

12.. Choi YH, Han MH, K OK, Craniofacial cavernous venous malformations: Percutaneous sclerotherapy with use of ethanolamine oleate: J Vasc Interv Radiol, 2002; 13(5); 475-82

13.. de Queiroz SB, de Lima VN, Amorim PH, Severe edema after sclero-therapy of labial hemangioma with ethamolin oleate in a young child: J Craniofac Surg, 2016; 27(6); e567-68

14.. Hedberg SE, Fowler DL, Ryan RL, Injection sclerotherapy of esophageal varices using ethanolamine oleate: Am J Surg, 1982; 143(4); 426-31

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16.. Fronek H, Fronek A, Saltzberg G, Allergic reactions to Sotradecol: J Dermatol Surg Oncol, 1989; 15(6); 684

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Figures

Figure 1.. (A) Hemangioma protruding from the vagina (white arrowhead); urethral meatus (yellow arrow); labia minora (white arrow). (B) Eruptive massive hemorrhage was noted with puncture of vaginal wall hemangioma (white arrowhead). (C) Hemoglobin (Hb) change during hospitalization and visits to previous physician.Figure 2.. The right vaginal wall is visible using a vaginal speculum. Bleeding from vaginal wall at 9 o’clock (white arrowhead).Figure 3.. (A) Dynamic computed tomography scan (axial) of the pelvic region in the arterial layer showing a strong contrast effect in the entire vagina, which is suggestive of abundant blood flow (white arrowheads). Urinary bladder (white arrow), rectum (yellow arrow). (B) Two months after embolization. Vaginal wall hemangioma is obscured (white arrowhead); urinary bladder (white arrow); rectum (yellow arrow). (C) Contrast-enhanced magnetic resonance image (T2, sagittal) of the pelvic region showing the entire vaginal wall seemed to be replaced by a hemangioma with a diameter of 10 cm (white arrowheads); urinary bladder (white arrow); rectum (yellow arrow). (D) Four months after sclerotherapy; vaginal wall hemangioma became obscured; uterus (white arrow); vagina (yellow arrow).Figure 4.. (A) Early layer of contrast injection to the right vaginal artery angiography. (B) Leakage of contrast medium observed from the peripheral blood vessels (white arrowhead), which was considered to be the cause of the bleeding.Figure 5.. (A) Transvaginal ultrasound probe with an egg collection needle attached. The egg collection needle protrudes from the tip (white arrowheads). (B) Monoethanolamine oleate is injected and hemangioma is contrasted (white arrowhead).

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923