Abstract

Background and methodology Vascular lesions of theuterus are rare but pose difficult management decisions.We reviewed the cases of four patients who presentedwith vascular uterine lesions following early pregnancyloss at the Royal Infirmary of Edinburgh, Edinburgh, UK in2006.

Results All four patients had signs on colour Dopplerimaging consistent with arteriovenous malformations andelevated serum human chorionic gonadotrophin (hCG)levels. In each case, once the hCG level had returned tonormal the uterine lesions resolved. The first case

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IntroductionVascular lesions are a rare finding when imaging isperformed for women who present with heavy vaginalbleeding and they pose difficult clinical managementdilemmas. They can represent a true arteriovenousmalformation (AVM), a haemangioma, gestationaltrophoblastic disease or retained products of conception.An AVM may be congenital, or acquired through pelvicsurgery, infection, trophoblastic disease, cervical orendometrial malignancy and trauma including curettage.1,2

Colour Doppler imaging is the preferred method ofdiagnosis, with angiography reserved for patients requiringtherapeutic embolisation.2,3 The recommended diagnosticsigns for an AVM are based on the presence of ahypervascular lesion with turbulent flow within themyometrium.1 However, these Doppler findings may alsobe observed in gestational trophoblastic disease and withretained products of conception (RPOC).1

Management of such lesions ranges from aconservative approach to invasive procedures includingembolisation or hysterectomy.

Report of casesOver a 1-year period, six patients with a vascular uterinelesion following early pregnancy failure in the firsttrimester were identified in the Simpson Centre forReproductive Health at the Royal Infirmary of Edinburgh,Edinburgh, UK. We searched the available literature foradvice on management but found a lack of clear guidelinesfor diagnosis or treatment. This prompted us to review the

Management of vascular uterine lesions associated withpersistent low-level human chorionic gonadotrophinKate L Darlow, Andrew W Horne, Hilary O D Critchley, Jane Walker, W Colin Duncan

SHORT COMMUNICATION

Simpson Centre for Reproductive Health, Royal Infirmary ofEdinburgh, Edinburgh, UKKate L Darlow, MBChB, Specialist Registrar

Department of Reproductive and Developmental Sciences,University of Edinburgh, UKAndrew W Horne, PhD, MRCOG, Principal LecturerHilary O D Critchley, MD, FRCOG, Professor of ReproductiveMedicineW Colin Duncan, MD, MRCOG, Consultant

Department of Radiology, Royal Infirmary of Edinburgh,Edinburgh, UKJane Walker, MBChB, FRCR, Consultant Radiologist

Correspondence to: Dr Kate Darlow, Department of Obstetricsand Gynaecology, St John’s Hospital, Howden, Livingston, WestLothian EH54 6PP, UK. E-mail: katedarlow@doctors.org.uk

required therapeutic embolisation but the subsequentpatients were successfully managed conservatively.

Conclusions Most uterine vascular lesions are attributedto arteriovenous malformations but those associated withpregnancy represent subinvolution of the placental bedand can be managed conservatively with monitoring ofhCG concentrations.

Keywords embolisation, haemorrhage, human chorionicgonadotrophin (hCG), miscarriage, vascular malformation

J Fam Plann Reprod Health Care 2008; 34(2): 118–120(Accepted 14 January 2008)

case records of the first four patients, which theninfluenced our clinical decisions for subsequent patients.

Case 1The first case was a 36-year-old nulliparous woman whopresented as an emergency with heavy vaginal bleeding, 8weeks’ amenorrhoea and a positive pregnancy test. She hada background of an irregular menstrual cycle sincestopping the oral contraceptive pill 18 months previously.

A pelvic ultrasound scan showed no evidence of anintrauterine pregnancy and the myometrium was of anormal texture. The patient’s serum human chorionicgonadotrophin (hCG) at that time was 81 IU/l. Theimpression was that the patient had had a completemiscarriage although an ectopic pregnancy could not beexcluded. She was therefore asked to return for a repeatserum hCG 48 hours later. The repeat hCG was 46 IU/l, and1 week later showed a slower fall than expected to 36 IU/l.

One week subsequently she returned with lowerabdominal pain, ongoing light vaginal bleeding and a low-grade pyrexia. A repeat hCG was 22 IU/l. She was admittedovernight and treated with intravenous antibiotics forsuspected endometritis. After a further 10 days she returnedwith a history of much heavier vaginal bleeding andcrampy abdominal pain. Her serum hCG was still 13 IU/l.A transvaginal ultrasound scan (TVS) showed an 8 × 6 × 11mm area of mixed echogenicity in the endometrium of theuterine fundus. In addition, the anterior myometrial wallcontained a highly vascular area, with both arterial andvenous flow on Doppler examination (Figure 1a). Theseappearances were suggestive of minimal RPOC and apossible uterine vascular malformation.

The management plan was for the patient to have arepeat TVS in 1 month. However, she returned as anemergency 3 weeks later, with further very heavy bleeding.Her haemoglobin fell from 119 to 95 g/l and her serumhCG was still raised at 8 IU/l. A repeat TVS showedincreasing vascular sinuses in the uterine wall. An AVM

Key message points� Conservative management of vascular uterine lesions is

appropriate when they are associated with elevatedhuman chorionic gonadotrophin.

� If such lesions are associated with very heavy bleeding,operative interference may be avoided by therapeuticembolisation.

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Management of vascular uterine lesions

was suspected and the patient was therefore taken forurgent radiological intervention. A computed tomography(CT) scan showed marked enhancement of the anterioruterine wall with contrast extravasation into the uterinecavity (Figure 1b). This was in keeping with activebleeding from a uterine vascular malformation. However,the lack of early venous filling and the appearance of thevessels during the angiography were not typical of AVM.She went on to undergo bilateral uterine arteryembolisation, which was successful in stemming the heavyvaginal bleeding.

Four months following embolisation the patient wasasymptomatic, having re-established a normal menstrualcycle. Her hCG was normal at <5 IU/l and a follow-upmagnetic resonance imaging (MRI) scan showed a normaluterus.

Case 2The second case was a 40-year-old woman who presentedwith a 3-year history of secondary infertility. She had had

one early miscarriage requiring surgical evacuation and aCaesarean section at term for transverse lie. She underwentroutine fertility investigations, including a TVS. The onlyabnormality found was a 61cm intramural fibroid at theuterine fundus, which did not distort the cavity. She thenbecame pregnant spontaneously but approximately 7 weeksfrom her last menstrual period she presented as anemergency with vaginal bleeding. TVS demonstrated fetaldemise and she underwent an uncomplicated surgicalevacuation with minimal blood loss.

One month later she was reviewed in our infertilityclinic and a TVS was performed to assess the fibroid. Thisremained unchanged, but a 3.7 × 5.3 cm area of mixedechogenicity, with an increased vascular signal, was notednear the cervix. At this time the patient was asymptomatic.However, her serum hCG was found to be elevated at 686IU/l. The possibility of a hydatidiform mole was raised andconsequently her hCG levels were monitored. As these fell,this diagnosis was thought unlikely and a plan was madefor conservative management. Over the following month

Figure 1 Imaging in vascular lesions. Case 1. (a) Transvaginal ultrasound scan showing a largely thin endometrial echo (E) with a highly vasculararea on colour flow Doppler (arrow) on the anterior uterine wall. (b) Computed tomography scan with contrast shows a brightly enhancingvascular serpiginous structure (arrow) extending from the uterine wall with extravasation into the uterine cavity. Case 2. (c) Magnetic resonanceimaging (MRI) scan of fibroid (F) and abnormal vascular area (arrow) above cervix (C). (d) Vascular enhanced MRI scan of uterus with increasedvascularity throughout the uterine wall (arrow) distorting the bladder (B)

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the patient remained asymptomatic and her hCG declinedslowly to 90 IU/l.

Before her next appointment, the patient presented asan emergency with heavy vaginal bleeding. Her serumhCG at this stage was slightly higher at 114 IU/l. A TVSshowed a 61cm mass of mixed echogenicity, whichappeared to extend from the uterine cavity anteriorly intoand through the myometrium. It was located low in thebody of the uterus, in the region of the previous Caesareansection scar. Colour Doppler imaging revealed the mass tobe highly vascular with low resistance flow. The cause ofthe bleeding at this time was uncertain but the differentialdiagnosis included RPOC, molar change and vascularmalformation and it was therefore important to establish atissue diagnosis.

Hysteroscopy showed a large red polyp-like structure,in continuity with the uterine wall, extending into thecavity. Minimal bleeding occurred during the procedure. Acareful biopsy was obtained. This showed inflamed andnecrotic decidua, but no chorionic villi or trophoblast.

As a result of the findings at hysteroscopy an MRIscan was performed (Figure 1c), which confirmed thepresence of a vascular lesion close to the cervix thatcrossed the myometrium and distorted the bladder (Figure1d). After discussion between gynaecology and radiologycolleagues it was decided that a conservative approachwas the best line of management. Over the followingweeks the patient was monitored as an outpatient. HerhCG levels gradually fell to normal and her menstrualcycle returned to a normal pattern. A repeat TVS at 3months showed a normal uterine cavity with completeresolution of the vascular lesion.

Cases 3 and 4The third patient was a 26-year-old woman who underwentsurgical management of an incomplete miscarriage at 12weeks and the fourth was a 24-year-old woman who hadundergone surgical termination of pregnancy at 10 weeks.Both women re-presented several weeks later with heavyvaginal bleeding and were found to have vascularmyometrial lesions on Colour Doppler imaging associatedwith elevated hCG levels. The vascular lesion in the fourthpatient increased in size (from 2.2 × 2.7 × 2.7 cm to 4.7 ×4.1 × 3.9 cm) and became more pronounced over the nextmonth with marked vascular sinuses. Both patients weremanaged conservatively, and within 2 to 3 months theirhCG levels had returned to normal and repeat TVSsshowed entirely normal uterine morphology.

DiscussionThis case series describes the management of four patientsfound to have vascular lesions of the uterus afterspontaneous miscarriage or first-trimester termination ofpregnancy.

With each patient, the colour Doppler imaging findingson TVS were consistent with the recognised signs for anAVM. However, angiography performed in the first case didnot show early venous contrast filling, which is the cardinalsign of AVM.3 Additionally, the spontaneous regression ofthe vascular lesions in our series is not typical of true AVMs.

Histopathological investigations have shown that AVMsoccur as a result of errors in morphogenesis. They arecomposed of stable endothelial cells, which therefore do notshow spontaneous regression.4

The presence of uterine vascular malformationsfollowing pregnancy has been reported by Timmerman etal. who felt that the condition may represent subinvolutionof the placental bed.3 Similarly, our impression is thatalthough the ultrasound imaging of our case seriesmimicked true AVMs, these lesions do representsubinvolution of retained placental tissue. By tracking thepatients’ hCG levels, which were all found to be elevatedand which resolved slowly as the vascular lesionsregressed, we have been able to support this hypothesis. Itis not known whether the trophoblast was karyotypicallynormal in these cases, but we do note the association withmiscarriage suggesting that there may be a link to abnormalplacentation or trophoblast function.

In the case report by Kido et al.5 a vascular lesion wasnoted within the uterus 6 weeks after surgical terminationof pregnancy. A hysterectomy was performed for asuspected AVM. However, pathology revealed onlynecrotic RPOC. These authors’ hypothesis was thatnecrosis of the chorionic villi left vessels witharteriovenous fistulas in the retained placental tissue. It wasonly noted retrospectively that serum hCG levels wereelevated and returned to normal postoperatively. As withthe first case in our series, intervention was necessary inview of the volume of bleeding. However, if this were nota factor, it is likely that the vascular lesion would haveresolved spontaneously.

ConclusionIt is appropriate that vascular uterine lesions following earlypregnancy loss are managed conservatively with hCGmonitoring, with the exception of patients who bleed heavilyand who are therefore at risk of vascular compromise, whomay be managed with therapeutic embolisation.

Statements on funding and competing interestsFunding None identified.Competing interests None identified.

References1 Mungen E. Vascular abnormalities of the uterus: have we

recently over-diagnosed them? Ultrasound Obstet Gynecol2003; 21: 529–531.

2 Timmerman D, Van den Bosch T, Peeraer K, Debrouwere E,Van Schoubroeck D, Stockx L, et al. Vascular malformations inthe uterus: ultrasonographic diagnosis and conservativemanagement. Eur J Obstet Gynecol Reprod Biol 2000; 92:171–178.

3 Timmerman D, Wauters J, Van Calenbergh S, VanSchoubroeck D, Maleux G, Van Den Bosch T, et al. ColorDoppler imaging is a valuable tool for the diagnosis andmanagement of uterine vascular malformations. UltrasoundObstet Gynecol 2003; 21: 570–577.

4 Mulliken JB, Glowacki J. Hemangiomas and vascularmalformations in infants and children: a classification based onendothelial characteristics. Plast Reconstr Surg 1982; 69:412–422.

5 Kido A, Togashi K, Koyama T, Ito H, Tatsumi K, Fujii S, et al.Retained products of conception masquerading as acquiredarteriovenous malformation. J Comput Assist Tomogr 2003; 27:88–92.

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