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Giant uterine artery pseudoaneurysm after a missed miscarriage
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termination in a cesarean scar pregnancy
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Yun Mou1, Yuezhen Xu2, Ying Hu1, Tianan Jiang1*
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1Department
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Zhejiang University, Hangzhou, P. R. China
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2Department
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*Corresponding author: Tianan Jiang
of Ultrasound, the First Affiliated Hospital, College of Medicine,
of Ultrasound, Zhejiang Xinan Hospital, Jiaxing, P.R. China
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Yun Mou: Department of Ultrasound, the First Affiliated Hospital, College of
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Medicine, Zhejiang University, Hangzhou, P. R. China (email address:
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xuyuntin@163.com)
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Postal address of the submitting author: 79# Qingchun Road, Hangzhou,
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310003, P.R. China
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Yuezhen Xu: Department of Ultrasound, Zhejiang Xinan Hospital, Jiaxing, P. R.
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China (email address: xuyuezhen001@sina.com)
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Ying Hu: Department of Ultrasound, the First Affiliated Hospital, College of
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Medicine, Zhejiang University, Hangzhou, P. R. China (email address:
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2216315256@qq.com)
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Corresponding author: Tianan Jiang, Department of Ultrasound, the First
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Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou, P. R.
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China (e-mail: chenmy69@126.com)
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Postal address: 79# Qingchun Road, Hangzhou, 310003, P.R. China (e-mail:
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chenmy69@126.com, Tel: 86-57 1-87236516; Fax: 86-571-87236514)
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Abstract
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Background
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Uterine artery pseudoaneurysms are dangerous and can lead to severe
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hemorrhage. We report an uncommon cause of a giant pseudoaneurysm in a
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missed miscarriage in a woman with a cesarean scar pregnancy.
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Case presentation
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The patient was a 25-year-old woman with a missed miscarriage in a cesarean
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scar pregnancy. Curettage was performed under ultrasound monitoring. The
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uterine artery pseudoaneurysm was detected on the next day by Doppler
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ultrasonography. It measured 71  44  39 mm. On day 10 after curettage, the
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pseudoaneurysm ruptured spontaneously and an emergency hysterectomy
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was performed.
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Conclusion
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Ultrasound and Doppler ultrasonography are recommended to rule out uterine
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artery pseudoaneurysms, especially in cases such as a cesarean scar
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pregnancy. For a giant uterine artery pseudoaneurysm, interventional
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embolization might be the first treatment option when the diagnosis is made,
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and, as in this case, hysterectomy is clearly possible when severe bleeding
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occurs.
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Keywords
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Uterus, Pseudoaneurysm, Ultrasound, Missed miscarriage, Cesarean scar
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pregnancy
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Background
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Uterine artery pseudoaneurysms are rare complications, which can arise after
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repeated curettage, abortions, cesarean sections, uncomplicated vaginal
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deliveries or reproductive tract infections. They are dangerous and can lead to
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severe hemorrhage. The interval from pelvic surgery to the onset of symptoms
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is typically 1 week to 3 months [1-4]. This delay is supposed to be caused by a
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gradual increase in the size of the pseudoaneurysm caused by a characteristic
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pressure increment. The blood flow into the pseudoaneurysm is greater during
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systole than diastole. This leads to a gradual pressure build up and eventual
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rupture. It can be treated with hysterectomy with or without hypogastric artery
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ligation. In recent years, uterine artery embolization has become an accepted
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treatment method for this condition. The option depends on the patient’s
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reproductive desires and hemodynamic situation.
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A literature search found three case reports of cesarean scar pregnancies
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complicated with a uterine artery pseudoaneurysm [5-7]. Here, it occurred in a
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patient with a missed miscarriage during a cesarean scar pregnancy and the
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lesion was the largest reported to date.
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Case presentation
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A 25-year-old woman had been amenorrheic for 2 months and was referred to
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the hospital because of painless vaginal bleeding that had lasted for 15 days.
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She had a history of one missed miscarriage at 9 weeks of gestation managed
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by surgical curettage 4 years before, and one elective cesarean delivery 2
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years before.
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gestational age, 20 days before presenting at the hospital, based on elevated
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urinary levels of beta human chorionic gonadotropin (-hCG). The vaginal
She had been diagnosed as being pregnant at 40 days of
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bleeding was scanty and her serum -hCG level was 1200 mIU/mL.
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Transvaginal ultrasonography showed that the uterus measured 106  64  60
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mm. There was an echo-free area above the inner cervical os, measuring 43 
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23 mm, without any blood flow signal, yolk sac or embryo present. A mixed
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echo mass measuring 29  15 mm was detected in the uterine cavity but no
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blood flow signal could be found in it. The patient was diagnosed as having
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had a missed miscarriage in the cesarean scar region of the uterus and
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curettage was performed with ultrasound monitoring. During the procedure,
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massive bleeding (~600 mL) occurred but this was stopped with an
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intravenous injection of oxytocin and uterine massage. Chorionic tissue was
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aspirated and proven as such by histopathology. When the curettage was
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finished, the uterine cavity was revealed as a clear thin line by ultrasound and
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was considered normal. Vaginal packing was performed subsequently. At 18
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hours after curettage, the serum -hCG level was 1164 mIU/mL. A cystic lesion
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with an uneven wall in the lower part of the uterus measuring 71  44  39 mm
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was detected with gray-scale ultrasonography (Fig. 1). Color Doppler
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ultrasonography showed a swirl of colors in the cystic lesion (Fig. 2). It was
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connected with an artery by a narrow neck in its posterior wall. The peak
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velocity in the artery was as high as 215 cm/s (Fig. 3). At day 10 after curettage,
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severe vaginal bleeding occurred suddenly and an emergency hysterectomy
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was performed. A ruptured pseudoaneurysm measuring 60  70  50 mm in
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the cesarean scar position of the uterus was found during the operation (Fig.4).
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The wall of the cyst was composed of clotted blood, decidual tissue and
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chorionic tissue.
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Discussion
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A uterine artery pseudoaneurysm is very dangerous and should be diagnosed
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as soon as possible. One of its causes is vascular injury following abortion,
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curettage, or pelvic surgery. Traumatic injury to the vessel wall causes wall
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incompetence
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Ultrasonography was useful in detecting the formation of the pseudoaneurysm
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in this case, both by 2-dimentional and Doppler scans. Using 2-dimentional
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ultrasonography, a pseudoaneurysm manifests as a hypoechoic mass and is
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thereby not easily differentiated from a hematoma or a true aneurysm. Color
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Doppler ultrasonography was helpful in this case as it demonstrated turbulent
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arterial flow with a to-and-fro pattern, connected to a parent artery by a narrow
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neck in the pseudoaneurysm. Blood flow into the mass during systole and
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away from the mass during diastole can be explained by the pressure gradient
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between a distended high-pressure pseudoaneurysm and the low pressure in
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the artery during diastole [8]. A true aneurysm manifests as a color-coded
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fusiform dilation of the parent artery and spectral analysis can demonstrate a
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typical arterial flow pattern. A simple hematoma does not reveal any color
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signal caused by turbulent blood flow. The wall of a pseudoaneurysm is formed
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by a peripheral thrombus. In this case, decidual tissue and chorionic tissue
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were also found in the wall. We diagnosed the pseudoaneurysm by
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ultrasonography at the day after curettage. Therefore, we think it is necessary
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for patients to undergo a Doppler ultrasound examination as a required
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postoperative investigation especially in cases of cesarean scar pregnancy.
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In this case, the pseudoaneurysm was located in the cesarean scar. The wall
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was very thin with a high risk of rupture. Endovascular treatment is often the
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first-line therapy. It can be achieved with embolization with coils, stents and
and
hemorrhage
leading
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to
a
pseudoaneurysm.
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injectable liquids [9, 10]. It offers the potential of preserving fertility for the
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patient. However, in the literature, the pseudoaneurysms treated successfully
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by this method were only 0.6–3.5 cm in diameter [11]. Our interventional
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radiologists lacked experience in treating such a giant lesion and thought that
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repeated treatment might be required. The serum -hCG level was still high
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after curettage, suggesting there were retained chorionic villi in the
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pseudoaneurysm. Recanalization of the pseudoaneurysm might occur even
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after arterial embolization from rapid recruitment of collateral vessels. In this
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situation, repeated uterine curettage immediately after embolization or
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methotrexate therapy might decrease the probability of recanalization.
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Therefore, in this case, the patient tried to seek admittance to an advanced
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institution to undergo embolization treatment. However, when waiting for this,
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massive bleeding occurred and we performed an emergency hysterectomy.
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Another possible treatment method was direct thrombin injection into the mass
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[12], but no further experience has been reported in the literature. We lack
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knowledge on the scope of possible complications, such as subsequent
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arterial thrombosis or allergic responses.
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Conclusions
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It is highly advisable for patients to undergo a Doppler ultrasound examination
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as a required postoperative investigation to rule out a uterine artery
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pseudoaneurysm, in cases of a cesarean scar pregnancy. For such a giant
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uterine artery pseudoaneurysm, embolization might be the first treatment when
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the diagnosis is made, but hysterectomy is possible when severe bleeding
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occurs.
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Consent
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Written informed consent was obtained from the patient for publication of this
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Case Report and the accompanying images. A copy of the written consent is
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available for review by the Editor of this journal.
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Competing interest
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The authors declare that they have no competing interests.
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Authors’ contributions
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YM carried out the ultrasonography, participated in treatment of the patient and
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wrote the manuscript. YX assisted with the ultrasonography and followed up
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the treatment of the patient. YH participated in the design of the study and
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helped to draft the manuscript. TJ conceived of the study and participated in
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the diagnosis and in drafting the report. All authors have read and approved
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the final manuscript.
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Acknowledgments
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This study was supported by the Zhejiang Provincial Natural Science
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Foundation, P. R. China (No LY13H180008), the National Natural Science
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Foundation, P. R. China (No 81371571) and the Education Agency of Zhejiang
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Province, P. R. China (No Y200907825).
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Figure legends
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Figure 1 2-dimentional ultrasound image of the pseudoaneurysm. It
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showed a pseudoaneurysm in the anterior wall of the lower part of the uterus.
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The wall varied in thickness and the inner side of the wall was uneven.
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Figure 2 Doppler ultrasonography of the pseudoaneurysm. A swirl of
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colors was seen in the color Doppler ultrasonography image. It showed the
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opening of the pseudoaneurysm and its supplying artery.
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Figure 3 Supplying artery velocity of the pseudoaneurysm. Pulsed
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Doppler ultrasonography showed that the velocity of blood in the supplying
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artery near the orifice of the pseudoaneurysm was as high as 215 cm/s.
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Figure 4 Gross specimen of the uterine artery pseudoaneurysm. The giant
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pseudoaneurysm (left) was ruptured and the size was consistent with the
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ultrasonography findings.
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