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Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper. The only source of funding for this project is the Department of Obstetrics and Gynecology at New York Presbyterian Hospital – Weill Cornell. The authors have no conflicts of interest to disclose.
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References Aide N, Deux J-F, Peretti I et al. 2005. Persistent foreign body reaction around inguinal mesh prostheses: a potential pitfall of FDG PET. American Journal of Roentgenology 184:1172–1177. Kitajima K, Murakami K, Yamasaki E et al. 2009. Performance of integrated FDGPET/contrast-enhanced CT in the diagnosis of recurrent uterine cancer: comparison with PET and enhanced CT. European Journal of Nuclear Medicine and Molecular Imaging 36:362–372. Nahabedian MY. 2009. AlloDerm performance in the setting of prosthetic breast surgery, infection, and irradiation. Plastic and Reconstructive Surgery 124:1743–1753. Orenstein SB, Qiao Y, Kaur M et al. 2010. Human monocyte activation by biologic and biodegradable meshes in vitro. Surgical Endoscopy 24:805–811. Park J-Y, Kim EN, Kim D-Y et al. 2008. Role of PET or PET/CT in the post-therapy surveillance of uterine sarcoma. Gynecologic Oncology 109:255–262. Rosenbaum SJ, Lind T, Antoch G et al. 2006. False-positive FDG PET uptake – the role of PET/CT. European Radiology 16:1054–1065. Shaltz AT, Leath CA, Wiedenhoefer JF et al. 2011. Reaction to a surgical implant foreign body masquerading as recurrent uterine sarcoma. Obstetrics and Gynecology 117:450–452. Yilmaz M, Sevinc A, Aybasti N et al. 2008. FDG uptake in abdominal mesh implant on FDG PET/CT. Clinical Nuclear Medicine 33:351–352.
Varied presentation of uterine arteriovenous malformations and their management by uterine artery embolisation N. Singh1, R. Tripathi1, Y. M. Mala1, S. Tyagi1, S. Tyagi2 & C. Singh1 Departments of 1Obstetrics and Gynaecology and 2Cardiology, Maulana Azad Medical College, New Delhi, India DOI: 10.3109/01443615.2013.816666 Correspondence: N. Singh, Department of Obstetrics and Gynaecology, Maulana Azad Medical College, New Delhi, India. E-mail: nilanchalisingh@ gmail.com
Uterine arteriovenous malformations are rare lesions which have a varied presentation that may range from the patient being asymptomatic to varying degrees of menorrhagia. It can be diagnosed by Doppler sonography but a strong index of suspicion is necessary. Management of this condition depends on presentation and available resources. In asymptomatic patients, we can leave the patients on regular follow-up as many lesions regress spontaneously. In patients with excessive haemorrhage not responding to embolisation or when facilities of embolisation are not available, hysterectomy needs to be done. Uterine artery embolisation should be considered the optimum treatment, as it has a high success rate coupled with few complications and also has fertility-preserving potential. Clinical follow-up usually suffices but sonography may occasionally be required.
aneurysm, arteriovenous fistula, arteriovenous aneurysm, pulsating angioma and cavernous angioma. They may be congenital or acquired. Congenital uterine AVMs arise from an abnormality in the embryological development of primitive vascular structures. Acquired uterine AVMs have been postulated to occur following surgical procedures, as they are frequently preceded by prior dilatation and curettage, uterine surgery or direct uterine trauma and are less commonly associated with endometrial carcinoma, cervical carcinoma or gestational trophoblastic disease, although the precise pathogenesis remains unclear (Ichikawa et al. 2003). Clinical suspicion is an essential prerequisite for diagnosis and treatment. This case series review reports four cases of arteriovenous malformations managed by uterine artery embolisation at the Department of Gynaecology and Cardiology, Maulana Azad Medical College, New Delhi, during May 2007 to March 2009.
Case reports Case 1 A 28-year-old female, P0A3 presented to the gynaecology outpatient’s department, with complaints of menorrhagia for 8 months. She had a history of spontaneous abortion followed by curettage 1 year previously. Her haemoglobin was 8.2 g% and transvaginal sonography showed an endometrial thickness of 5 mm with normal adnexa (Figure 1). There were anechoic spaces present throughout the myometrium, which were more concentrated in the area of the uterine fundus as seen on MRI and this led to suspicion of uterine arteriovenous malformation (Figure 2). Her Doppler sonography showed colour aliasing with apparent flow reversals and pulsatile high velocity waveforms in the myometrium (Figure 3). Magnetic resonance angiography was also done for confirmation, which showed arteriovenous malformation involving both the uterine arteries and the myometrial vasculature with feeders from the internal iliac artery, together with early opacification of the right common iliac vein and the inferior vena cava (Figure 4). With the diagnosis of uterine arteriovenous malformation being confirmed, the patient was scheduled for uterine artery embolisation. The patient was discharged after the procedure and she remained asymptomatic during her 2-year follow-up.
Case 2 A 38-year-old patient, P3A1, reported to casualty with complaints of torrential bleeding per vaginam for 1 hour, which had been preceded by spotting for a few hours; there was no previous history of amenorrhoea. Her previous cycles had been regular with increased bleeding during some cycles. The patient was very pale. Her clothes were soaked with blood and she had signs of hypovolaemia. Her systemic examination was normal. Pelvic examination revealed that there was bleeding through the os and the uterus was bulky and uniformly enlarged, firm, mobile and non-tender with no palpable adnexa. Her urinary pregnancy test was negative. She had a report of
Keywords: Arteriovenous malformation, Doppler sonography, menorrhagia, uterine artery embolisation
Introduction Uterine arteriovenous malformations (AVMs) are rare lesions (3–4.5% cases of menorrhagia) (O’Brien et al. 2006). Arteriovenous malformation is known by various names, e.g. cirsoid
Figure 1. Transvaginal sonography of Case 1, depicting anechoic spaces in the uterine myometrium.
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Figure 2. Sagittal T2W magnetic resonance image of Case 1, showing multiple flow void in the myometrium with disruption of junctional anatomy. The endometrium is normal.
trans-abdominal ultrasonography done 3 days prior, in view of menorrhagia, which showed no uterine pathology and normal endometrial thickness. Immediate resuscitative measures were instituted and she was transfused three units of blood. Uterine tamponade was done using a Foley’s catheter, following which the haemorrhage was controlled. Colour Doppler examination revealed a large uterine arteriovenous malformation involving the whole of the uterine myometrium. She was scheduled for uterine artery embolisation, which was performed the next day. The bleeding was controlled following the procedure and she was subsequently discharged on haematinics. She remained asymptomatic during her 18 months follow-up, with normal menstrual cycles.
ing the parametrium and the adnexal regions. The MR angiogram image showed the uterine arteriovenous malformation with feeders from the internal iliac artery and early opacification of the right common iliac vein and the inferior vena cava. These findings confirmed the diagnosis of uterine AVM. Her uterine artery embolisation was subsequently performed on a day-care basis and she was asymptomatic during her 1 year follow-up.
Case 3
Case 4
A 44-year-old patient, P4, who had previously had a salpingectomy, presented to the outpatient’s department with complaint of menorrhagia for the past 6 months. She had been previously diagnosed and managed as a case of dysfunctional uterine bleeding. Earlier records of transabdominal ultrasonography showed no pelvic pathology and endometrial aspiration histology showed normal secretory endometrium. The patient had taken progesterones as advised by her physician but menorrhagia had persisted and she was referred. Transvaginal sonography (TVS) was performed to rule out any submucous fibroid and the endometrial cavity appeared normal. A Doppler examination was also done. This confirmed the suspicion of uterine AVM but there was evidence of intense vascularity in the entire pelvic region, so an MRI and MR angiography were performed. These revealed a bulky uterine fundus with multiple tortuous flow-related signal voids within the myometrium. The flow voids were also seen involv-
A 22-year-old woman, P1A1, had a missed abortion with partial expulsion of the fetus, which was followed by curettage. The patient continued to bleed moderately for 1 month. She went to a private hospital where a repeat curettage was done, as incomplete abortion was suspected. She continued to bleed after the curettage and hence was investigated for persistent trophoblastic disease but that possibility was eliminated as the serum β-hCG was only 22.4 IU/ml and a transvaginal scan showed normal endometrial thickness and normal adnexa. She was further evaluated by Doppler sonography, which showed findings suggestive of AVM, i.e. anechoic, tortuous, tubular and serpiginous spaces within the myometrium, with intense colour fill within the anechoic spaces. Uterine artery embolisation was done but the patient continued to have menorrhagia for 3 months and was referred for repeat embolisation. At this time, the pelvic examination was normal and a repeat Doppler evaluation was done, which showed persistence of the uterine arteriovenous malformation but the flow pattern showed smaller sized vessels with less vascularity compared with the previous scan. In view of these findings, a decision was taken for hormonal therapy rather than perpetuating the risk of a repeat embolisation. She was kept under close surveillance and started on oral medroxyprogesterone, 10 mg t.i.d., for 3 cycles. She responded well on the hormone therapy and was also subsequently eumenorrhoeic at the 15-month follow-up.
Figure 4. Magnetic resonance angiography of Case 1, showing uterine arteriovenous malformation with feeders from internal iliac artery.
Discussion
Figure 3. Doppler sonography of Case 1, showing intense colour filling within anechoic uterine spaces and evidence of colour aliasing. Aliasing is bright and turbulent flow in Doppler, which is indicative of high velocity blood flow.
Uterine AVMs are rare lesions reported in women aged 18–72 years (Arora et al. 2004). In this series, the patients’ ages varied from 22 to 44 years. Two patients were multiparous and one of these had an earlier salpingectomy and the other was using barrier contraception; one was nulliparous and desired a pregnancy, while the last was symptomatic post-abortion. Three of these four patients had a history of curettage, which is considered to be the most common predisposing factor leading to uterine AVM formation.
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The clinical presentation may vary, from no symptoms to a varying degree of menorrhagia (Elia et al. 2001). Patients may also present with postpartum or post-abortal haemorrhage. Metrorrhagia is a frequent presentation at emergency, often requiring curettage but in undiagnosed cases of AVM of the uterus, curettage may lead to massive and fatal haemorrhage due to direct vascular trauma (Dorez et al. 2010). In our study, three patients presented with menorrhagia and one of them had an episode of massive, vaginal bleeding. One patient presented with post-abortal haemorrhage. In the past, diagnosis of AVM was difficult and was made by using invasive diagnostic techniques, such as contrast enhanced angiography. Some cases were diagnosed after laparotomy, whereas others were diagnosed only after histopathology of a hysterectomy specimen (Grivell et al. 2005). With the advances in diagnostic equipment, such as colour Doppler sonography; contrast enhanced computed tomography (CT); magnetic resonance imaging (MRI) and magnetic resonance angiography (MRA), however, their detection has become easier and today, AVMs are being detected even in asymptomatic patients (Huang et al. 1998). This has increased the frequency of detection, although it is still difficult to comment on the actual increase of prevalence rates. Gray-scale sonography may show an inhomogeneous myometrium with hypoechogenic tubular structures. These findings are nonspecific and, at times, inconspicuous and difficult to note (Maldonado et al. 2008). Colour Doppler imaging shows uterine hypoechoic or anechoic spaces with high velocity vascular network in these cases. It may also show multiple extensive vascular channels in the parametrium. There may be a tangle of vessels with multidirectional highvelocity flow producing a ‘colour mosaic’ pattern. Spectral Doppler analysis reveals arteriovenous shunting with high-velocity, low-resistance flow (O’Brien et al. 2006). The systolic and diastolic velocities in these malformations may be 4–6 times higher than that observed in normal myometrial vessels. Resistance index values range between 0.25 and 0.55 and pulsatility index values range from 0.3 to 0.6 (AlShekaili et al. 2011). MRI scans may reveal the presence of multiple tortuous flow voids, suggesting abnormal vessels in the myometrium and adnexa. Angiographic features consist of a complex tangle of vessels supplied by enlarged feeders from the uterine arteries (Verma et al. 2008) and occasionally the ovarian artery. Arterial computed tomography and MRA may confirm the diagnosis and also identify the supplying vessel (Dorez et al. 2010). In our study, the diagnosis was made by colour Doppler in all cases, though in two patients, MRI and MRA were performed to confirm the diagnosis. Colour and pulse-wave Doppler along with angiography, confirm the diagnosis and gives important information regarding the size and exact location of the lesion. These facts may help the interventional radiologist and treating physician to decide whether embolisation or hysterectomy is required (Gillis and Davie 2012). It is important to emphasise that management depends on clinical presentation and not the ultrasonographic findings alone, especially in the current era of increasing frequency of diagnoses (O’Brien et al. 2006). Conservative management should be considered in clinically stable patients, since most of these lesions tend to resolve spontaneously (Timmerman et al. 2000; Flynn and Levine 1996; Beller et al. 1988). In the past, uterine AVMs have been treated by uterine artery ligation or hysterectomy but today, hysterectomy is only performed when fertility preservation is not required, there is limited access to medical facilities or when embolisation therapy fails. AVM can also be managed successfully by ligation of the feeding artery (Yokomine et al. 2008). Laparoscopic bipolar coagulation of the uterine blood vessels can also be performed (Wu et al. 2001). Currently, uterine artery embolisation has revolutionised the management of uterine AVMs and is preferred because it is minimally invasive and has fertility-preserving potential. The decision for performing embolisation in a patient with uterine AVM should not depend on the size of the AVM but should be based on the patient’s clinical status. Women with a single episode of bleeding who are haemodynamically stable can be treated expectantly or with one or more of the available drugs. These include oestrogens, progestins, methylergonovine, danazol and 15-methyl-prostaglandin F2α (Ghosh et al. 1986; Elia et al. 2001). In patients with recurrent bleeding episodes or severe degrees of
menorrhagia, a more definitive therapy is warranted and embolisation can be considered. In this series, three patients had severe degrees of menorrhagia and had uterine artery embolisation. They all responded to the procedure and were asymptomatic during their follow-up period, which varied from 12 to 24 months. One patient had excessive bleeding following surgical evacuation for incomplete abortion and she also underwent uterine artery embolisation but her bleeding continued despite the procedures. It was then decided to put her on oral progesterones for a few months and she responded to this combination therapy. Yang and co-workers (2005) reported the long-term success rate of embolisation for uterine AVMs as 79% (11 of 14 patients) and Kwon and Kim (2002) reported a 90% success rate (22 of 24 patients). The overall efficacy was reported as 93% by Ghai and co-workers (2003), in a retrospective review of 15 patients over 10 years. A total of 25 embolisation procedures were performed in these 15 patients; six required repeat embolisation (one patient underwent embolisation as many as six times and was pregnant when the series was reported) and five underwent embolisation twice for recurrence of bleeding. Only one patient underwent a hysterectomy. No patient required repeat embolisation in the four patients reported in our series. This may be due to the fact that newer equipment with better resolution is likely to make a significant contribution to optimal placement of embolic device. It is also possible that if combination therapy is considered (e.g. hormonal together with embolisation, as in our last case), the need for repeat procedures may be minimised. This may be considered worthwhile, as complications are likely to escalate with increasing numbers of repeat procedures. It is also possible that not all patients show an immediate response and the use of supportive measures in the interim period may result in eventual success. The recent developments of newer materials for embolisation, coupled with improved image resolution and radiation safety measures, would further contribute to the greater use and acceptability of embolisation. The selection of embolic material depends on the size of the AVMs and choice of interventional radiologist. These may be absorbable gelatin sponge, stainless steel coils or particles of polyvinyl alcohol, the absorbable gelatin sponge being considered an ideal material. In this series, the material used for embolisation was PVA particles. There are studies reporting selective embolisation of the feeding vessel for treatment of uterine AVM; successful treatment outcome in a patient of secondary postpartum haemorrhage due to AVM was reported after the selective embolisation of the feeding vessel (Nanjundan et al. 2011). Selective embolisation with successful outcome has been reported in cornual pregnancy associated with uterine AVM (Valsky et al. 2007). Some authors recommend elective internal iliac angiography on the affected side and evaluating the contralateral arteries after unilateral uterine artery embolisation, so that the inconspicuous feeding arteries may then be identified and embolised if required (Maldonado et al. 2008). One case (Bagga et al. 2008) reported selective embolisation of the feeder of AVM but the patient became symptomatic after 6 months and a hysterectomy had to be performed. In another case report, there was selective embolisation of the feeding uterine artery but later, to ensure complete cessation of supply to AVM, another uterine artery was also embolised (Castillo et al. 2007). In many other studies (e.g. Kwon and Kim 2002; O’Brien et al. 2006), bilateral uterine artery embolisation was done, as in all the four cases we reported. Bilateral uterine artery embolisation may prevent the anastomotic vessels developing from the non-feeding vessel to cause recurrence of the lesion. Hence, we recommend bilateral uterine artery embolotherapy, especially in extensive lesions because they usually have bilateral vascular supply. Varying degrees of pelvic pain are common in the immediate postembolisation period. Some patients may have acute and/or chronic pain, temperatures of up to 102° Fahrenheit, malaise, nausea, vomiting and severe night sweats, collectively known as the post-embolisation syndrome (PES). Serious complications occur mainly due to internal iliac artery embolisation or the use of glue as the embolic agent or both. These complications include perianal skin sloughing,
Gynaecology Case Reports 107 uterovaginal and rectovaginal fistulas and neurological deficits in the lower extremities but these complications are rare. There were no serious complications in our series of patients. Patients in this case series had follow-up periods varying from 8 to 12 months and remained eumenorrhoeic during this period. Follow-up of patients undergoing uterine artery ligation should be done clinically as well as by endovaginal sonography (O’Brien et al. 2006). High success rates have been reported in the short term but only long-term follow-up will determine the true success rate of these procedures as longterm follow-up after an apparently successful embolisation may reveal more failures. The cause of failure of embolisation is still not clear and may depend upon the type of embolic material used and the extent of vascular recanalisation, together with the expertise of the intervention radiologist or regrowth of the AVMs due to inherent factors.
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Conclusion Endovaginal sonography, together with colour Doppler imaging is the investigative modality of choice to diagnose uterine AVM and should be considered in those patients where the cause of menorrhagia is considered to be hormonal but there is no response to conventional medical or hormonal therapy. To reiterate, uterine artery embolisation should be considered the optimum treatment, as it has a high success rate coupled with few complications, and also has fertility preserving potential. Clinical follow-up usually suffices but sonography may occasionally be required. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Al-Shekaili KR, Bhatnagar G, Ramadhan FA, Al-Zadjali N. 2011. Arteriovenous malformation of uterus. Indian Journal of Pathology and Microbiology 54:187–188. Arora R, Batra A, Saxena P, Gupta P, Minocha B. 2004. Arteriovenous malformations of the uterus. New Zealand Medical Journal 117:1206. Bagga R, Verma P, Aggarwal N, Suri V, Bapuraj JR, Kalra N. 2008. Failed angiographic embolization in uterine arteriovenous malformation: a case report and review of the literature. Medscape Journal of Medicine 10:12. Beller U, Rosen RJ, Beckan EM, Markoff G, Berenstein A. 1988. Congenital arteriovenous malformation of the female pelvis: a gynecologic perspective. American Journal of Obstetrics and Gynecology 159:1153–1160. Castillo MS, Borge MA, Pierce KL. 2007. Embolization of a traumatic uterine arteriovenous malformation. Seminars in Interventional Radiology 24:296–299. Dorez M, Delotte J, Novellas S, Chevallier P, Bongain A. 2010. Uterine arterio-venous malformation. International Journal of Emergency Medicine 3:505–506. Elia G, Counsell C, Singer SJ. 2001. Uterine artery malformation as a hidden cause of severe uterine bleeding; a case report. Journal of Reproductive Medicine 46:398–400. Flynn MK, Levine D. 1996. The noninvasive diagnosis and management of arteriovenous malformation. Obstetrics and Gynecology 88:650–652. Ghai S, Rajan DK, Asch MR, Muradali D, Simons ME, TerBrugge KG. 2003. Efficacy of embolization in traumatic uterine vascular malformations. Journal of Vascular and Interventional Radiology 14:1401–1408. Ghosh TK. 1986. Arteriovenous malformation of the uterus and pelvis. Obstetrics and Gynecology 68:40S–43S. Gillis K, Davie K. 2012. Sister vascular anomalies in the nongravid uterus arteriovenous malformation versus arteriovenous fistula. Journal of Diagnostic Medical Sonography 28:135–139. Grivell M, Reid M, Mellor A. 2005. Uterine arteriovenous malformations: a review of current literature. Obstetrical and Gynecological Survey 60:761–767. Huang MW, Muradali D, Thurston WA, Burns PN, Wilson SR. 1998. Uterine arteriovenous malformations: gray scale and Doppler ultrasound features with MR imaging correlation. Radiology 206:115–123. Ichikawa Y, Nakauchi T, Sato T, Oki A, Tsunoda H, Yoshikawa H. 2003. Ultrasound diagnosis of uterine arteriovenous fistula associated with placental site trophoblastic tumor. Ultrasound in Obstetrics and Gynecology 21:606–608. Kwon JH, Kim GS. 2002. Obstetric iatrogenic arterial injuries of the uterus: diagnosis with US and treatment with transcatheter arterial embolization. Radiographics 22:35–46.
Maldonado J, Perez C, Rodriguez W. 2008. AJR Teaching file: profuse vaginal bleeding seven weeks following induced abortion. American Journal of Roentgenology191: S79–S82. Nanjundan P, Rohilla M, Raveendran A, Jain V, Khandelwal N. 2011. Pseudoaneurysm of uterine artery: a rare cause of secondary postpartum hemorrhage, managed with uterine artery embolisation. Journal of Clinical Imaging Science 1:14. O’Brien P, Neyastani A, Buckley AR, Chang SD, Legiehn GM. 2006. Uterine arteriovenous malformations: from diagnosis to treatment. Journal of Ultrasound in Medicine 25:1387–1392. Timmerman D, Van den Bosch T, Peeraer K, Debrouwere E, Van Schoubroeck D, Stockx L et al. 2000. Vascular malformation in the uterus :ultrasonographic diagnosis and conservative management. European Journal of Obstetrics, Gynecology, and Reproductive Biology 92:171–178. Valsky DV, Hamani Y, Verstandig A, Yagel S. 2007. The use of 3D rendering, VCI-C, 3D power Doppler and B-flow in the evaluation of interstitial pregnancy with arteriovenous malformation treated by selective uterine artery embolization. Ultrasound in Obstetrics and Gynecology 29:352–355. Verma A, Mohan S, Chandra T, Kathuria MK, Baijal SS. 2008. Percutaneous angio-embolization of a post laparoscopy complex utero-adnexal vascular malformation. Journal of Minimal Access Surgery 4:48–50. Wu YC, Liu WM, Yuan CC, Ng HT. 2001. Successful treatment of symptomatic arteriovenous malformation of the uterus using laparoscopic bipolar coagulation of uterine vessels. Fertility and Sterility 76:1270–1271. Yang JJ, Xiang Y, Wan XR, Yang XY. 2005. Diagnosis and management of uterine arteriovenous fistulas with massive vaginal bleeding. International Journal of Gynecology and Obstetrics 89:114–119. Yokomine D, Yoshinaga M, Baba Y, Mastuo T, Iguro Y, Nakajo M et al. 2009. Successful management of arteriovenous malformation by ligation of feeding artery after unsuccessful uterine artery embolization. Journal of Obstetrics and Gynaecology Research 35:183–188.
Massive ascites caused by a large pedunculated subserosal uterine leiomyoma that has feeding arteries from peripheral tissues and exhibits elevated CA125: A case report of atypical Pseudo-Meigs’ syndrome H.-K. Yip1, L.-W. Huang1,2,3, Y.-H. Lin1,2 & J.-L. Hwang1,3 1Department of Obstetrics and Gynecology, Shin Kong Wu Ho-Su
Memorial Hospital, Taipei, 2School of Medicine, Fu-Jen Catholic University, New Taipei City and 3School of Medicine, Taipei Medical University, Taipei, Taiwan DOI: 10.3109/01443615.2013.832736 Correspondence: L-W. Huang, Department of Obstetrics and Gynecology, Shin Kong Wu Ho-Su Memorial Hospital, No. 95, Wen Chang Road, Shih Lin District, Taipei City 111, Taiwan. E-mail: [email protected]
Introduction Leiomyoma is a common gynaecological disease, the incidence of which increases with age; 4.3 per 1,000 woman-years for 25–29-yearolds and 22.5 for 40–44-year-olds. Subserosal leiomyoma includes types that are at least 50% intramural, less than 50% intramural or are attached to the serosa by a stalk; so-called pedunculated subserosal leiomyoma (Parker 2012). Pedunculated subserosal leiomyomas develop on an extended stalk from the uterus or may even be separate from the uterus, with a vascular connection to the pelvic wall or other intra-abdominal site. The latter are parasitic leiomyoma. Symptoms at presentation cover a wide range (Rader et al. 1990; Fasih et al. 2008). We report a case of pedunculated subserosal leiomyoma associated with massive ascites and an elevated level of CA125.
Case report A 41-year-old woman, gravid 2, para 2, presented with abdominal fullness and prolonged menstrual periods for 3 years. Physical examination showed abdominal distension with shifting dullness. Ultrasound of
Gynaecology Case Reports
Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper. The only source of funding for this project is the Department of Obstetrics and Gynecology at New York Presbyterian Hospital – Weill Cornell. The authors have no conflicts of interest to disclose.
J Obstet Gynaecol Downloaded from informahealthcare.com by University of Aberdeen on 04/25/14 For personal use only.
References Aide N, Deux J-F, Peretti I et al. 2005. Persistent foreign body reaction around inguinal mesh prostheses: a potential pitfall of FDG PET. American Journal of Roentgenology 184:1172–1177. Kitajima K, Murakami K, Yamasaki E et al. 2009. Performance of integrated FDGPET/contrast-enhanced CT in the diagnosis of recurrent uterine cancer: comparison with PET and enhanced CT. European Journal of Nuclear Medicine and Molecular Imaging 36:362–372. Nahabedian MY. 2009. AlloDerm performance in the setting of prosthetic breast surgery, infection, and irradiation. Plastic and Reconstructive Surgery 124:1743–1753. Orenstein SB, Qiao Y, Kaur M et al. 2010. Human monocyte activation by biologic and biodegradable meshes in vitro. Surgical Endoscopy 24:805–811. Park J-Y, Kim EN, Kim D-Y et al. 2008. Role of PET or PET/CT in the post-therapy surveillance of uterine sarcoma. Gynecologic Oncology 109:255–262. Rosenbaum SJ, Lind T, Antoch G et al. 2006. False-positive FDG PET uptake – the role of PET/CT. European Radiology 16:1054–1065. Shaltz AT, Leath CA, Wiedenhoefer JF et al. 2011. Reaction to a surgical implant foreign body masquerading as recurrent uterine sarcoma. Obstetrics and Gynecology 117:450–452. Yilmaz M, Sevinc A, Aybasti N et al. 2008. FDG uptake in abdominal mesh implant on FDG PET/CT. Clinical Nuclear Medicine 33:351–352.
Varied presentation of uterine arteriovenous malformations and their management by uterine artery embolisation N. Singh1, R. Tripathi1, Y. M. Mala1, S. Tyagi1, S. Tyagi2 & C. Singh1 Departments of 1Obstetrics and Gynaecology and 2Cardiology, Maulana Azad Medical College, New Delhi, India DOI: 10.3109/01443615.2013.816666 Correspondence: N. Singh, Department of Obstetrics and Gynaecology, Maulana Azad Medical College, New Delhi, India. E-mail: nilanchalisingh@ gmail.com
Uterine arteriovenous malformations are rare lesions which have a varied presentation that may range from the patient being asymptomatic to varying degrees of menorrhagia. It can be diagnosed by Doppler sonography but a strong index of suspicion is necessary. Management of this condition depends on presentation and available resources. In asymptomatic patients, we can leave the patients on regular follow-up as many lesions regress spontaneously. In patients with excessive haemorrhage not responding to embolisation or when facilities of embolisation are not available, hysterectomy needs to be done. Uterine artery embolisation should be considered the optimum treatment, as it has a high success rate coupled with few complications and also has fertility-preserving potential. Clinical follow-up usually suffices but sonography may occasionally be required.
aneurysm, arteriovenous fistula, arteriovenous aneurysm, pulsating angioma and cavernous angioma. They may be congenital or acquired. Congenital uterine AVMs arise from an abnormality in the embryological development of primitive vascular structures. Acquired uterine AVMs have been postulated to occur following surgical procedures, as they are frequently preceded by prior dilatation and curettage, uterine surgery or direct uterine trauma and are less commonly associated with endometrial carcinoma, cervical carcinoma or gestational trophoblastic disease, although the precise pathogenesis remains unclear (Ichikawa et al. 2003). Clinical suspicion is an essential prerequisite for diagnosis and treatment. This case series review reports four cases of arteriovenous malformations managed by uterine artery embolisation at the Department of Gynaecology and Cardiology, Maulana Azad Medical College, New Delhi, during May 2007 to March 2009.
Case reports Case 1 A 28-year-old female, P0A3 presented to the gynaecology outpatient’s department, with complaints of menorrhagia for 8 months. She had a history of spontaneous abortion followed by curettage 1 year previously. Her haemoglobin was 8.2 g% and transvaginal sonography showed an endometrial thickness of 5 mm with normal adnexa (Figure 1). There were anechoic spaces present throughout the myometrium, which were more concentrated in the area of the uterine fundus as seen on MRI and this led to suspicion of uterine arteriovenous malformation (Figure 2). Her Doppler sonography showed colour aliasing with apparent flow reversals and pulsatile high velocity waveforms in the myometrium (Figure 3). Magnetic resonance angiography was also done for confirmation, which showed arteriovenous malformation involving both the uterine arteries and the myometrial vasculature with feeders from the internal iliac artery, together with early opacification of the right common iliac vein and the inferior vena cava (Figure 4). With the diagnosis of uterine arteriovenous malformation being confirmed, the patient was scheduled for uterine artery embolisation. The patient was discharged after the procedure and she remained asymptomatic during her 2-year follow-up.
Case 2 A 38-year-old patient, P3A1, reported to casualty with complaints of torrential bleeding per vaginam for 1 hour, which had been preceded by spotting for a few hours; there was no previous history of amenorrhoea. Her previous cycles had been regular with increased bleeding during some cycles. The patient was very pale. Her clothes were soaked with blood and she had signs of hypovolaemia. Her systemic examination was normal. Pelvic examination revealed that there was bleeding through the os and the uterus was bulky and uniformly enlarged, firm, mobile and non-tender with no palpable adnexa. Her urinary pregnancy test was negative. She had a report of
Keywords: Arteriovenous malformation, Doppler sonography, menorrhagia, uterine artery embolisation
Introduction Uterine arteriovenous malformations (AVMs) are rare lesions (3–4.5% cases of menorrhagia) (O’Brien et al. 2006). Arteriovenous malformation is known by various names, e.g. cirsoid
Figure 1. Transvaginal sonography of Case 1, depicting anechoic spaces in the uterine myometrium.
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Figure 2. Sagittal T2W magnetic resonance image of Case 1, showing multiple flow void in the myometrium with disruption of junctional anatomy. The endometrium is normal.
trans-abdominal ultrasonography done 3 days prior, in view of menorrhagia, which showed no uterine pathology and normal endometrial thickness. Immediate resuscitative measures were instituted and she was transfused three units of blood. Uterine tamponade was done using a Foley’s catheter, following which the haemorrhage was controlled. Colour Doppler examination revealed a large uterine arteriovenous malformation involving the whole of the uterine myometrium. She was scheduled for uterine artery embolisation, which was performed the next day. The bleeding was controlled following the procedure and she was subsequently discharged on haematinics. She remained asymptomatic during her 18 months follow-up, with normal menstrual cycles.
ing the parametrium and the adnexal regions. The MR angiogram image showed the uterine arteriovenous malformation with feeders from the internal iliac artery and early opacification of the right common iliac vein and the inferior vena cava. These findings confirmed the diagnosis of uterine AVM. Her uterine artery embolisation was subsequently performed on a day-care basis and she was asymptomatic during her 1 year follow-up.
Case 3
Case 4
A 44-year-old patient, P4, who had previously had a salpingectomy, presented to the outpatient’s department with complaint of menorrhagia for the past 6 months. She had been previously diagnosed and managed as a case of dysfunctional uterine bleeding. Earlier records of transabdominal ultrasonography showed no pelvic pathology and endometrial aspiration histology showed normal secretory endometrium. The patient had taken progesterones as advised by her physician but menorrhagia had persisted and she was referred. Transvaginal sonography (TVS) was performed to rule out any submucous fibroid and the endometrial cavity appeared normal. A Doppler examination was also done. This confirmed the suspicion of uterine AVM but there was evidence of intense vascularity in the entire pelvic region, so an MRI and MR angiography were performed. These revealed a bulky uterine fundus with multiple tortuous flow-related signal voids within the myometrium. The flow voids were also seen involv-
A 22-year-old woman, P1A1, had a missed abortion with partial expulsion of the fetus, which was followed by curettage. The patient continued to bleed moderately for 1 month. She went to a private hospital where a repeat curettage was done, as incomplete abortion was suspected. She continued to bleed after the curettage and hence was investigated for persistent trophoblastic disease but that possibility was eliminated as the serum β-hCG was only 22.4 IU/ml and a transvaginal scan showed normal endometrial thickness and normal adnexa. She was further evaluated by Doppler sonography, which showed findings suggestive of AVM, i.e. anechoic, tortuous, tubular and serpiginous spaces within the myometrium, with intense colour fill within the anechoic spaces. Uterine artery embolisation was done but the patient continued to have menorrhagia for 3 months and was referred for repeat embolisation. At this time, the pelvic examination was normal and a repeat Doppler evaluation was done, which showed persistence of the uterine arteriovenous malformation but the flow pattern showed smaller sized vessels with less vascularity compared with the previous scan. In view of these findings, a decision was taken for hormonal therapy rather than perpetuating the risk of a repeat embolisation. She was kept under close surveillance and started on oral medroxyprogesterone, 10 mg t.i.d., for 3 cycles. She responded well on the hormone therapy and was also subsequently eumenorrhoeic at the 15-month follow-up.
Figure 4. Magnetic resonance angiography of Case 1, showing uterine arteriovenous malformation with feeders from internal iliac artery.
Discussion
Figure 3. Doppler sonography of Case 1, showing intense colour filling within anechoic uterine spaces and evidence of colour aliasing. Aliasing is bright and turbulent flow in Doppler, which is indicative of high velocity blood flow.
Uterine AVMs are rare lesions reported in women aged 18–72 years (Arora et al. 2004). In this series, the patients’ ages varied from 22 to 44 years. Two patients were multiparous and one of these had an earlier salpingectomy and the other was using barrier contraception; one was nulliparous and desired a pregnancy, while the last was symptomatic post-abortion. Three of these four patients had a history of curettage, which is considered to be the most common predisposing factor leading to uterine AVM formation.
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Gynaecology Case Reports
The clinical presentation may vary, from no symptoms to a varying degree of menorrhagia (Elia et al. 2001). Patients may also present with postpartum or post-abortal haemorrhage. Metrorrhagia is a frequent presentation at emergency, often requiring curettage but in undiagnosed cases of AVM of the uterus, curettage may lead to massive and fatal haemorrhage due to direct vascular trauma (Dorez et al. 2010). In our study, three patients presented with menorrhagia and one of them had an episode of massive, vaginal bleeding. One patient presented with post-abortal haemorrhage. In the past, diagnosis of AVM was difficult and was made by using invasive diagnostic techniques, such as contrast enhanced angiography. Some cases were diagnosed after laparotomy, whereas others were diagnosed only after histopathology of a hysterectomy specimen (Grivell et al. 2005). With the advances in diagnostic equipment, such as colour Doppler sonography; contrast enhanced computed tomography (CT); magnetic resonance imaging (MRI) and magnetic resonance angiography (MRA), however, their detection has become easier and today, AVMs are being detected even in asymptomatic patients (Huang et al. 1998). This has increased the frequency of detection, although it is still difficult to comment on the actual increase of prevalence rates. Gray-scale sonography may show an inhomogeneous myometrium with hypoechogenic tubular structures. These findings are nonspecific and, at times, inconspicuous and difficult to note (Maldonado et al. 2008). Colour Doppler imaging shows uterine hypoechoic or anechoic spaces with high velocity vascular network in these cases. It may also show multiple extensive vascular channels in the parametrium. There may be a tangle of vessels with multidirectional highvelocity flow producing a ‘colour mosaic’ pattern. Spectral Doppler analysis reveals arteriovenous shunting with high-velocity, low-resistance flow (O’Brien et al. 2006). The systolic and diastolic velocities in these malformations may be 4–6 times higher than that observed in normal myometrial vessels. Resistance index values range between 0.25 and 0.55 and pulsatility index values range from 0.3 to 0.6 (AlShekaili et al. 2011). MRI scans may reveal the presence of multiple tortuous flow voids, suggesting abnormal vessels in the myometrium and adnexa. Angiographic features consist of a complex tangle of vessels supplied by enlarged feeders from the uterine arteries (Verma et al. 2008) and occasionally the ovarian artery. Arterial computed tomography and MRA may confirm the diagnosis and also identify the supplying vessel (Dorez et al. 2010). In our study, the diagnosis was made by colour Doppler in all cases, though in two patients, MRI and MRA were performed to confirm the diagnosis. Colour and pulse-wave Doppler along with angiography, confirm the diagnosis and gives important information regarding the size and exact location of the lesion. These facts may help the interventional radiologist and treating physician to decide whether embolisation or hysterectomy is required (Gillis and Davie 2012). It is important to emphasise that management depends on clinical presentation and not the ultrasonographic findings alone, especially in the current era of increasing frequency of diagnoses (O’Brien et al. 2006). Conservative management should be considered in clinically stable patients, since most of these lesions tend to resolve spontaneously (Timmerman et al. 2000; Flynn and Levine 1996; Beller et al. 1988). In the past, uterine AVMs have been treated by uterine artery ligation or hysterectomy but today, hysterectomy is only performed when fertility preservation is not required, there is limited access to medical facilities or when embolisation therapy fails. AVM can also be managed successfully by ligation of the feeding artery (Yokomine et al. 2008). Laparoscopic bipolar coagulation of the uterine blood vessels can also be performed (Wu et al. 2001). Currently, uterine artery embolisation has revolutionised the management of uterine AVMs and is preferred because it is minimally invasive and has fertility-preserving potential. The decision for performing embolisation in a patient with uterine AVM should not depend on the size of the AVM but should be based on the patient’s clinical status. Women with a single episode of bleeding who are haemodynamically stable can be treated expectantly or with one or more of the available drugs. These include oestrogens, progestins, methylergonovine, danazol and 15-methyl-prostaglandin F2α (Ghosh et al. 1986; Elia et al. 2001). In patients with recurrent bleeding episodes or severe degrees of
menorrhagia, a more definitive therapy is warranted and embolisation can be considered. In this series, three patients had severe degrees of menorrhagia and had uterine artery embolisation. They all responded to the procedure and were asymptomatic during their follow-up period, which varied from 12 to 24 months. One patient had excessive bleeding following surgical evacuation for incomplete abortion and she also underwent uterine artery embolisation but her bleeding continued despite the procedures. It was then decided to put her on oral progesterones for a few months and she responded to this combination therapy. Yang and co-workers (2005) reported the long-term success rate of embolisation for uterine AVMs as 79% (11 of 14 patients) and Kwon and Kim (2002) reported a 90% success rate (22 of 24 patients). The overall efficacy was reported as 93% by Ghai and co-workers (2003), in a retrospective review of 15 patients over 10 years. A total of 25 embolisation procedures were performed in these 15 patients; six required repeat embolisation (one patient underwent embolisation as many as six times and was pregnant when the series was reported) and five underwent embolisation twice for recurrence of bleeding. Only one patient underwent a hysterectomy. No patient required repeat embolisation in the four patients reported in our series. This may be due to the fact that newer equipment with better resolution is likely to make a significant contribution to optimal placement of embolic device. It is also possible that if combination therapy is considered (e.g. hormonal together with embolisation, as in our last case), the need for repeat procedures may be minimised. This may be considered worthwhile, as complications are likely to escalate with increasing numbers of repeat procedures. It is also possible that not all patients show an immediate response and the use of supportive measures in the interim period may result in eventual success. The recent developments of newer materials for embolisation, coupled with improved image resolution and radiation safety measures, would further contribute to the greater use and acceptability of embolisation. The selection of embolic material depends on the size of the AVMs and choice of interventional radiologist. These may be absorbable gelatin sponge, stainless steel coils or particles of polyvinyl alcohol, the absorbable gelatin sponge being considered an ideal material. In this series, the material used for embolisation was PVA particles. There are studies reporting selective embolisation of the feeding vessel for treatment of uterine AVM; successful treatment outcome in a patient of secondary postpartum haemorrhage due to AVM was reported after the selective embolisation of the feeding vessel (Nanjundan et al. 2011). Selective embolisation with successful outcome has been reported in cornual pregnancy associated with uterine AVM (Valsky et al. 2007). Some authors recommend elective internal iliac angiography on the affected side and evaluating the contralateral arteries after unilateral uterine artery embolisation, so that the inconspicuous feeding arteries may then be identified and embolised if required (Maldonado et al. 2008). One case (Bagga et al. 2008) reported selective embolisation of the feeder of AVM but the patient became symptomatic after 6 months and a hysterectomy had to be performed. In another case report, there was selective embolisation of the feeding uterine artery but later, to ensure complete cessation of supply to AVM, another uterine artery was also embolised (Castillo et al. 2007). In many other studies (e.g. Kwon and Kim 2002; O’Brien et al. 2006), bilateral uterine artery embolisation was done, as in all the four cases we reported. Bilateral uterine artery embolisation may prevent the anastomotic vessels developing from the non-feeding vessel to cause recurrence of the lesion. Hence, we recommend bilateral uterine artery embolotherapy, especially in extensive lesions because they usually have bilateral vascular supply. Varying degrees of pelvic pain are common in the immediate postembolisation period. Some patients may have acute and/or chronic pain, temperatures of up to 102° Fahrenheit, malaise, nausea, vomiting and severe night sweats, collectively known as the post-embolisation syndrome (PES). Serious complications occur mainly due to internal iliac artery embolisation or the use of glue as the embolic agent or both. These complications include perianal skin sloughing,
Gynaecology Case Reports 107 uterovaginal and rectovaginal fistulas and neurological deficits in the lower extremities but these complications are rare. There were no serious complications in our series of patients. Patients in this case series had follow-up periods varying from 8 to 12 months and remained eumenorrhoeic during this period. Follow-up of patients undergoing uterine artery ligation should be done clinically as well as by endovaginal sonography (O’Brien et al. 2006). High success rates have been reported in the short term but only long-term follow-up will determine the true success rate of these procedures as longterm follow-up after an apparently successful embolisation may reveal more failures. The cause of failure of embolisation is still not clear and may depend upon the type of embolic material used and the extent of vascular recanalisation, together with the expertise of the intervention radiologist or regrowth of the AVMs due to inherent factors.
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Conclusion Endovaginal sonography, together with colour Doppler imaging is the investigative modality of choice to diagnose uterine AVM and should be considered in those patients where the cause of menorrhagia is considered to be hormonal but there is no response to conventional medical or hormonal therapy. To reiterate, uterine artery embolisation should be considered the optimum treatment, as it has a high success rate coupled with few complications, and also has fertility preserving potential. Clinical follow-up usually suffices but sonography may occasionally be required. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
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Massive ascites caused by a large pedunculated subserosal uterine leiomyoma that has feeding arteries from peripheral tissues and exhibits elevated CA125: A case report of atypical Pseudo-Meigs’ syndrome H.-K. Yip1, L.-W. Huang1,2,3, Y.-H. Lin1,2 & J.-L. Hwang1,3 1Department of Obstetrics and Gynecology, Shin Kong Wu Ho-Su
Memorial Hospital, Taipei, 2School of Medicine, Fu-Jen Catholic University, New Taipei City and 3School of Medicine, Taipei Medical University, Taipei, Taiwan DOI: 10.3109/01443615.2013.832736 Correspondence: L-W. Huang, Department of Obstetrics and Gynecology, Shin Kong Wu Ho-Su Memorial Hospital, No. 95, Wen Chang Road, Shih Lin District, Taipei City 111, Taiwan. E-mail: [email protected]
Introduction Leiomyoma is a common gynaecological disease, the incidence of which increases with age; 4.3 per 1,000 woman-years for 25–29-yearolds and 22.5 for 40–44-year-olds. Subserosal leiomyoma includes types that are at least 50% intramural, less than 50% intramural or are attached to the serosa by a stalk; so-called pedunculated subserosal leiomyoma (Parker 2012). Pedunculated subserosal leiomyomas develop on an extended stalk from the uterus or may even be separate from the uterus, with a vascular connection to the pelvic wall or other intra-abdominal site. The latter are parasitic leiomyoma. Symptoms at presentation cover a wide range (Rader et al. 1990; Fasih et al. 2008). We report a case of pedunculated subserosal leiomyoma associated with massive ascites and an elevated level of CA125.
Case report A 41-year-old woman, gravid 2, para 2, presented with abdominal fullness and prolonged menstrual periods for 3 years. Physical examination showed abdominal distension with shifting dullness. Ultrasound of
