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doi:10.1111/jog.12402

J. Obstet. Gynaecol. Res. Vol. 40, No. 6: 1791–1794, June 2014

Spontaneous rupture of uterine varicose veins: A rare cause for obstetric shock Pei Shan Lim1, Soon Pheng Ng2, Mohammad Nasir Shafiee1, Nirmala Kampan1 and Muhammad Abdul Jamil1 1

Department of O&G, UKMMC, Kuala Lumpur, and 2Columbia Asia Hospital, Puchong, Selangor, Malaysia

Abstract Spontaneous rupture of uterine surface varicose veins is rare but may result in serious complication of pregnancy, as it is associated with high perinatal and maternal mortality. We report a 24-year-old primigravida who presented with this rare condition mimicking placenta abruption, which was successfully managed. A review of reported cases was performed. Key words: hemoperitoneum, obstetric shock, uterine varicose veins, utero-ovarian veins.

Introduction Spontaneous rupture of uterine surface varicose veins is rare but may result in serious complication of pregnancy. It is associated with high perinatal and maternal mortalities. We report this rare condition, mimicking placenta abruption, which was successfully managed in our center.

Case Report A 24-year old primigravida presented in labor at 37 weeks of gestation. She was diagnosed to be in latent phase of labor but developed sudden onset of generalized abdominal pain 2 h later. There was no vaginal bleeding, history of trauma or motor vehicle accident. She was found to be pale with signs of shock. Her pulse rate was 110 b.p.m. and blood pressure was 90/60 mmHg. There was generalized tenderness per abdomen and the cervical os was 1 cm dilated. The cardiotocograph showed persistent fetal tachycardia. A diagnosis of placental abruption with concealed hemorrhage was made. She was resuscitated with i.v. fluid and prepared for an emergency cesarean section.

Intraoperatively, approximately 1500 mL of hemoperitoneum was noted upon entry into the abdomen. A live, female infant weighing 1730 g was delivered and did not require admission to the neonatal unit. The Apgar scores were 5 and 9 at 1 and 5 min, respectively. The placenta was delivered by controlled cord traction and there was no clinical evidence of abruption such as blood stained liquor or retroplacental clot. The uterus was exteriorized, without difficulty, following delivery of placenta to facilitate the search for source of bleeding. Multiple, dilated, thin-walled vessels coursing over the left lateral wall of the posterior aspect of the uterine surface were seen. A network of varicose vessels had ruptured through the serosal surface (Fig. 1). Hemostasis was secured by use of hot pack compression and hemostatic sutures. She received a total of 3 units of whole blood, 1 unit intraoperatively and another 2 units postoperatively. Both mother and baby were discharged well on the third postoperative day.

Discussion Uterine venous rupture is a very rare condition, which can lead to intra-abdominal hemorrhage. There were

Received: July 24 2013. Accepted: January 11 2014. Reprint request to: Dr Pei Shan Lim, Department of O&G, UKMMC, Jalan Yaakob Latif, 56000 Cheras, Kuala Lumpur, Malaysia. Email: [email protected]; [email protected]

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

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Figure 1 Network of varicose vessels that had ruptured through the serosal surface.

15 cases reported in the published work in the last decade. The etiology of these venous ruptures is unknown but few possible causes have been postulated. These include a sudden rise in venous pressure of the varicose, hypertrophied utero-ovarian veins; the critical point is reached especially during contraction and while straining in labor.1 Chronic inflammation secondary to endometriosis, a potential etiology, leading to friable vessels that may easily rupture, has also been suggested as a possible cause2 which was not seen in our case. Attaining a correct preoperative diagnosis is highly unlikely due to its rarity. As in most cases, this patient presented with sudden onset of abdominal pain in the third trimester in the absence of vaginal bleeding. A working clinical diagnosis of abruption placenta was made in view of maternal hypotension and fetal tachycardia. This is the commonest condition to be considered when a patient presents with painful contractions, hypotension and fetal distress. Other differential diagnoses include uterine rupture, intestinal obstruction, acute appendicitis and rarely ruptured splenic/hepatic artery. As it was an obstetric emergency with the working diagnosis of abruption and

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fetal distress in a laboring mother at 37 weeks of gestation, immediate resuscitation and prompt cesarean delivery minimized maternal and perinatal morbidity or mortality. Although bedside ultrasound scan is a readily available imaging modality in the labor room, it plays a limited role in the management of acute placental abruption, particularly in the presence of fetal compromise. Apart from demonstrating the presence of either retroplacental clot or hemoperitoneum in this case, a bedside ultrasound scan would not alter the management. However, in a less critical, subacute or doubtful situation, especially in preterm pregnancy, a highresolution color Doppler ultrasound scan coupled with a skillful sonographer may facilitate the diagnosis of bleeding, large, dilated, uterine varix from a chronic abruption as demonstrated by Kusanovic et al. In their case report, a prenatal diagnosis of bleeding cervical varix was made by transvaginal color Doppler ultrasound in the second trimester.3 In the absence of an obvious cause for the hemoperitoneum, a careful search should be made for the source of bleeding which includes inspection of the posterior uterine surface and the ovarian plexus. Other conditions that may give rise to similar clinical and intraoperative features include uterine arteriovenous malformation and uterine hemangioma have been reported.4,5 Confirmatory diagnosis may be possible following angiographic assessment or histological examination on hysterectomy specimen. Most uterine venous bleeding can be arrested following application of hemostatic sutures and compression. Nonetheless, hysterectomy had been reported as a last resort.6 A review of case reports available in the English language up till the last decade was performed (Table 1). There were five reports on bleeding secondary to spontaneous rupture of varicose veins on the uterine surface and three reports on bleeding secondary to spontaneous rupture of utero-ovarian vessels. All these cases occurred in the third trimester. However, there was one case reported by Ziereisen et al. where the patient presented on the third day postdelivery.12 The majority of these women had no active vaginal bleeding but developed shock together with severe abdominal pain. Urgent abdominal deliveries were mostly decided for suspected uterine/scar rupture, abruption placenta or hemoperitoneum of unknown reason. All were successfully managed by hemostatic sutures or by compression with the exception of one case that needed hysterectomy and one case

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

Rupture of uterine varicose veins

Table 1 Review of cases being reported Cole et al., 2005 33 years old G2P1 40 weeks POA

7

Hashimoto et al., 20066 40 years old G2

Moreira et al., 20098 39 years old G3P2 40 weeks POA

Zhang et al., 20099 38 years old G1P0 29 weeks POA IVF Twin pregnancy Cerclage performed

History

Examination/investigations

Findings/management

Gestational thrombocytopenia Previous CS Laparoscopic cystectomy for endometrioma In labor 8 h later, left shoulder pain Fainting spell Generalized abdominal pain Nausea, vomiting, diarrhea

Vital sign stable on admission Os 3 cm BP 80/50 Fetal bradycardia Os 3 cm

EM CS for suspected scar rupture EBL 3 L Bleeding from left utero-ovarian vessels (distorted area due to previous laparoscopic procedure) Admitted to ICU for 4 days Discharge day 6

Frequent uterine contraction with prolonged FHR deceleration after 4 h

Bilateral inguinal hernia repair Admitted in labor Had vacuum-assisted delivery after 5 h Bwt 3.68 kg A/S 9/10 Bilateral shoulder discomfort during second stage and episiotomy repair Slight vaginal bleeding Respiratory difficulty 8 h after delivery, complained of severe abdominal pain History of endometriosis, laparoscopic adhesiolysis and cystectomy Acute abdominal pain, nausea, vomiting No vaginal bleeding Subsequently developed upper abdominal pain

Hemodynamically instable Massive peritoneal free fluid noted upon ultrasound scan BP 70/25 1 cm cervical tear at 12 o’clock repaired but symptom persisted Hb 9.1 g/dL BP 101/60 Hb 8.7 g/dL

EM CS for suspected abruption Hemoperitoneum 500 mL Bleeding from varicose vein on posterior uterine surface Hysterectomy was performed Bwt 1.73 kg A/S 5/9 Laparotomy performed Laceration of anterior leaf of broad ligament, active bleeding from utero-ovarian vessels Hemoperitoneum 3000 mL Successfully managed by utero-ovarian vessel ligation

Zhang et al., 20098 35 years old G1P0 35 weeks POA IVF

History of laparoscopic adhesiolysis for endometriosis Sudden and intense right upper abdominal pain No vaginal bleeding No contraction

Zhang et al., 20099 34 years old G2P1 30 weeks POA IVF

Previous CS Previous salpingostomy Abdominal pain radiating to the back for 10 h No vaginal bleeding

Giulini et al., 201010 31 years old G4P1 33 weeks POA

Diffuse abdominal pain Nausea, vomiting No trauma No uterine contraction No vaginal bleeding

Nakaya et al., 201111 25 years old G1P0 28 weeks POA

Severe right lateral abdominal pain

Normal vital signs and FHR on admission Abdominal guarded, rebound tenderness Tender uterus with contraction, Os closed Became pale, with fetal bradycardia Progressive anemia CT scan suggestive of hemoperitoneum Abdominal paracentesis: blood Febrile BP 90/75, PR 100 Generalized abdominal tenderness Uterine irritable Normal FHR Progressive anemia and shock BP low, fast PR Abdomen guarded Tender uterus Non-reactive CTG Progressive anemia U/S showed free fluid BP 90/60 Tender uterus Uterus corresponding to gestation age Os closed, no vaginal bleeding Ultrasound scan: FHR positive, placenta posterior Progressive anemia CTG: low variability Presence of contraction Tocolysis

EM CS for suspected uterine rupture Two dead fetuses Ruptured uterine varix on posterior surface of the right uterine fundus EBL 3100 mL Discharge day 10

EM CS Hemoperitoneum 1.7 L Bleeding from fragile veins at right side of uterus Bwt 2.58 kg A/S 9/9

EM CS for suspected placental abruption Hemoperitoneum 1.5 L Bleeding from fragile vein at left cornu Bwt 1.075 kg A/S 4/7 needed ventilation Hemostasis sutures EM CS for suspected acute abdomen due to hemoperitoneum Pfannenstiel incision Large hemoperitoneum Bleeding from varix on posterior wall of left broad ligament Hemostasis suture 4 units of packed cell and 1 unit of FFP EBL 2.5–3 L Bwt 2.11 kg A/S 3/7 Discharge day 7 EM CS for non-reassuring CTG Hemoperitoneum 850 mL Bleeding from ruptured superficial varicose vein (right lateral portion of the uterus): compression and oxidized cellulose cotton Bwt 1.14 kg

A/S, Apgar score; Bwt, birthweight; CTG, cardiotocography; EBL, estimated blood loss; EM CS, emergency cesarean section; G, gravida; FFP, fresh frozen plasma; FHR, fetal heart rate; Hb, hemoglobin; ICU, intensive care unit; P, para; POA, period of amenorrhea; U/S, ultrasound scan.

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

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that needed unilateral oophorectomy. The majority had a favorable perinatal outcome but a case with twin pregnancy ended with perinatal death. Most of the cases presented in a dramatic manner and prompt action was subsequently instituted. Hence, there was no maternal mortality reported in these eight case reports as compared to earlier reports. Hemoperitoneum secondary to spontaneous rupture of uterine surface varicose veins should be considered as one of the differential diagnoses for obstetric shock, especially with the symptom of sudden abdominal pain in the absence of revealed vaginal bleeding, after careful exclusion of other common obstetric conditions. If the usual diagnosis is excluded, careful inspection and search for the primary source of bleeding should be performed. These include inspection of the posterior surface of the uterus and ovarian plexus to look for spontaneous rupture of varicose veins as illustrated in this case. Hemostatic sutures and direct compression are effective methods in arresting the bleeding, although in rare situations a hysterectomy may be needed to control bleeding.

Disclosure We declare that we have no conflict of interest.

References 1. Hodgkinson C, Christensen R. Hemorrhage from ruptured utero-ovarian veins during pregnancy; report of 3 cases and review of the literature. Am J Obstet Gynecol 1950; 59: 1112– 1117.

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2. Inoue T, Moriwaki T, Niki I. Endometriosis and spontaneous rupture of utero-ovarian vessels during pregnancy. Lancet 1992; 340: 240–241. 3. Kusanovic JP, Soto E, Espinoza J et al. Cervical varix as a cause of vaginal bleeding during pregnancy: Prenatal diagnosis by color Doppler ultrasonography. J Ultrasound Med 2006; 25: 545–549. 4. Castillo MS, Borge MA, Pierce KL. Embolization of a traumatic uterine arteriovenous malformation. Semin Intervent Radiol 2007; 24: 296–299. 5. Chou WY, Chang HW. Uterine hemangioma. Arch Pathol Lab Med 2012; 136: 567–571. 6. Hashimoto K, Tabata C, Ueno Y, Fukuda H, Shimoya K, Murata Y. Spontaneous rupture of uterine surface varicose veins in pregnancy: A case report. J Reprod Med 2006; 51: 722–724. 7. Cole M, Elton C, Bosio P, Waugh JJS. Spontaneous rupture of utero-ovarian vessels in labour – A rare case of obstetric haemoperitoneum. J Obstet Gynaecol 2005; 25: 301–303. 8. Moreira A, Reynolds A, Baptista P, Costa AR, Bernardes J. Case report: Intra-partum utero-ovarian vessels rupture. Arch Gynecol Obstet 2009; 279: 583–585. 9. Zhang Y, Zhao Y, Wei Y, Li R, Qiao J. Spontaneous rupture of subserous uterine veins during late pregnancy after in vitro fertilization. Fertil Steril 2009; 92: 395.e13–e16. 10. Giulini S, Zanin R, Volpe A. Hemoperitonuem in pregnancy from a ruptured varix of broad ligament. Arch Gynecol Obstet 2010; 282: 459–461. 11. Nakaya Y, Itoh H, Muramatsu K et al. A case of spontaneous rupture of a uterine superficial varicose vein in midgestation. J Obstet Gynecol Res 2011; 37: 1149–1153. 12. Ziereisen V, Bellens B, Gerard C, Baeyens L. Spontaneous rupture of utero-ovarian vessels in the postpartum period: Report of a case and literature review. J Gynecol Obstet Biol Reprod (Paris) 2003; 32: 51–54.

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

Spontaneous rupture of uterine varicose veins: a rare cause for obstetric shock.

Spontaneous rupture of uterine surface varicose veins is rare but may result in serious complication of pregnancy, as it is associated with high perin...
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