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A Rare Case of Bilateral Congenital A V Malformation

Introduction

Persistent or intermitted menorrhagia is documented as a clinical sign for AV malformation. The AVM lesions present a potential risk to patients. AVM is prone to deficient diagnosis (1). This is due to AVM clinical presentation that varies from no clinical signs to different levels of menorrhagia (2). The clinical presentation could manifest through life-threatening vaginal bleeding hence predispose anaemia.

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AVM diagnosis requires the highest level of clinical suspicion if appropriate medical intervention namely conservative or surgical would be conducted and implemented (1). The patient ought to be appropriately assessed for cases of abnormal or heavy uterine bleeding. Accuracy of AVM diagnosis helps to provide opportunities for appropriate treatment to be carried out which facilitates in eliminating opportunities for total abdominal hysterectomy in women that would prefer to have their reproductive system maintained after education on methods of intervention that could be implemented and their outcomes and obtaining informed consent from the patient (2).

Diagnosis of AVM requires multiple methods like hysteroscopy, Doppler flow, ultrasound and pelvic angiography (2). A uterine AVM lesion has different medical descriptions ranging from crisoid aneurysm, arteriovenous fistule, arteriovenous aneurysm, pulsating angioma and cavernous angioma. The earlier methods of diagnosis involved angiography, laprotomy or pathology (3). The clinical findings using earlier methods of AVM assessment were not reliable since uterine curettage was not theraupetic and had probabilities of aggravating the AVM. Hormonal therapy may not provide an alternative method for the management of AVM.

Advancement in technological development has resulted in the use of color Doppler sonography, contrast computed tomography (CT) and use of Magnetic Resonance Imaging (MRI). Advancement in technology has made it possible to detect cases of minor AVM (4).

This study reports on a rare case of bilateral congenital uterine AVM that was not sufficiently diagnosed due to a variation of clinical signs that were presented. This report emphasis on consideration of bilateral AVM pre-assessment and the use of multiple approaches to the diagnosis of AVM as a sustainable measure for identification of Unilateral or bilateral AVM and determination of site and size of AVM when developing treatment modalities in order to improve health outcomes.

Case Report

Patient Details

S.G., 23 years, GRAVIDA 4, PARA 1+3, having had two surgical TOPS and one medical TOP.

S.G. AVM Case and development

The woman, 23 years old with GRAVIDA 4, PARA 1+3 was admitted following gynecology assessment after referral from A&E. The woman previously had two surgical TOPS and one medical TOP at a private clinic. The woman bleeding was observed to be heavy and was admitted for further observation. Blood test showed Hb 13.3, Wcc of 8.5 and a blood group O, rhesus negative. Upon performance of ultrasound scan, the woman was observed to have normal anteverted uterus that had endometrial thickness of 11mm. It had minimal retained products. A clot was however observed in the left adnexa that measured 6.3 by 6.2cm. The woman had solid and cystic component and high vascular flow on Doppler. The woman Bhc was 10iu/l. There was no free fluid that was evident on the pelvis. The woman was given misoprostol 400mg and discharged.

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Four weeks later, the woman was re-admitted from A&E with severe vaginal bleeding. Pregnancy tests were negative. Her Bhc was less than 2. It was estimated that the woman. had lost about 3000mls of blood. The woman had two incidents of cardiac arrest in the A&E. after intervention radiologist review, pelvic angiography was carried out. This showed a large pelvic AVM on the right side of uterus and was embolised. Vaginal bleeding stopped and the woman was discharged on a combined oral contraceptives.

Two weeks later, the woman was re-admitted from A&E with a severe episode of vaginal bleeding. It was estimated the woman had lost about 4000mls of blood. Hb was determined to 6mg/l. The woman was taken to theatre for emergency laparotomy and bilateral internal iliac artery ligation. It was noted intraoperatively that there was a large pulsating mass in the left broad ligament that measured 6cm. there was evidence of uterine perforation in the supracervical region on the left side. There was however no evidence of intraperitoneal bleeding at the time of the operation. The woman management involved 10 units of blood transfusion, 4 units FFP and 3 units of platelets. This resulted into stopping of bleeding and the patient was taken to ITU for further observation and management.

Two days into the ITU, The woman started to bleed heavily and lost about 4000mls of blood. The woman was taken back to the theatre. Intervention radiologist found collaterals from the aorta to the both sides of the uterine AVM. Originally, the radiologist had embolized the right hand side. An emergency total abdominal hysterectomy was carried out. The ovaries were retained though. There was no further evidence of bleeding.

Discussion

Review of the literature

Arteriovenous malformation (AVM) is characterized by a proliferation of arterial and venous channels. AVM could present fistula formation. There could be evidence of small capillary like channels. AVM results into failure to identify or differentiate between the vein and the artery. This is secondary to initial thickening that is subject to intraluminal pressure. The acquired AVM results from hysterectomy, pelvic trauma, previous pregnancy or possible abortion, gestational trophoblastic illness, exposure of a female to diethyl stilbesterol, endometriosis, fibromyoma, endometrial or cervical cancer, previous use of intrauterine contraceptive devices and previous uterine surgery like curettages, caesarian section delivery.

From the patient presentation and case development, the woman had a rare bilateral congenital uterine AV malformation. This was subject to lack of differentiation of arteries and veins (5). There were multiple presences of anastomoses amongst the muscles, skin and viscera. The AVM was aggravated by the woman previous two surgical TOPS and one medical TOP. The surgeries were secondary to the woman AV malformation. The case for the woman was rare.

There was no clinical presentation for AVM (2). This is because initial speculum examination showed normal vulva, vagina and cervix. Ultrasound scan shown normal anteverted uterus that had endometrial thickness of 11mm. The woman didn’t show any possibilities of retained products (7). Doppler tests however showed a large complex mass in the left adnexa that measured 6.3 by 6.2cm respectively. The woman presented a high vascular flow on Doppler.

This resulted into implementation of conservative management methods where the woman was given misoprostol 400mg and discharged. The major presenting problem that the woman presented was heavy bleeding that was responsible for her observed two cardiac arrests while the woman was in the A&E (4). The woman case of bilateral AV malformation was not supported by the woman possible evidence of previous heavy bleeding or possibility of persistent abortion or possible immediate post abortion curettages. The woman case didn’t provide evidence of possible endometrium becoming exposed after shedding (5).

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The heavy bleeding in many cases of AVM that contributes to readmission is based on blood vessel erosion or when endometrium gets exposed post shedding. The woman was diagnosed with uterine perforations in supracervical region on the left side. This was suspected to have been the primary cause for the woman intermitted and persistent heavy bleeding since the woman tested negative for pregnancy (6).

Pelvic angiography on the woman identified a large pelvic AV malformation that was on the right hand side of the uterus and embolized (2). This made bleeding to settle and the woman was given combined contraceptive pills as part of management care plan. Persistence of bleeding two weeks later contributed into decreased Hb to 6gm/l from initial Hb 13.3. The woman underwent laparotomy and bilateral internal iliac artery ligation. The woman intraoperatively was observed to have had a large pulsating mass in the left broad ligament that measured 6 cm.

The woman had evidence of uterine perforation in supracervical region on the left side. The woman post operative management involved 10 units of blood transfusion, 4 units of FFP and 3 units of blood platelets. Post transfer to ITU for further management demonstrated the laparotomy and bilateral internal iliac artery ligation was not the primary cause of bleeding. The woman continued to bleed and underwent emergency total abdominal hysterectomy. The woman ovaries were retained for reproduction purposes (7). This followed radiology intervention that has identified collaterals from the aorta to both sides of the AV malformation. This confirmed the woman had bilateral AV malformation.

The woman AV malformation case highlights the importance of exhaustive assessment with regard to bilateral AV malformation (6). It also demonstrates the responsibility of utilization of investigational technology in diagnosis of AV malformation. The woman congenital AVM was managed through intra arterial embolization. This meant emergency laparotomy and bilateral internal iliac artery ligation was not a sustainable solution to the woman AVM. Total abdominal hysterectomy provided the woman congenital AVM management (7). The case demonstrates that uterine angiography and transvaginal ultrasonography should be used in diagnosis of congenital AV malformation.

The case demonstrates embolization with particulate matter subject to haemorrhagic conditions should be utilized in AVM management. Embolization substances should be used singly or in combination. The case demonstrated that arteriovenous shunting plays a vital role in heavy uterine bleeding. This affects haemodynamic outcomes. The heavy bleeding as observed in the woman has effect of contributing into haemorrhagic shock as the woman presented in the A&E.

The case further demonstrates that uterine congenital AVM in the event a patient presents refractory bleeding (4). The case determined that it is important to assess and evaluate possible future impacts of curettage, parturition and surgery. The case determined that vascular embolization through use of either ultrasoniography or angiography should be integrated into armamentarium. This should be carried out as a remedy for managing severe uterine bleeding that has relationships with AV malformation (6, 7). The case further determined that when a patient fails to present audible bruit or pulsatile masses are not felt at vaginal examination, then colour Doppler sonography should be considered as alternative method for congenital AVM diagnosis and assessment.

Conclusion

The study outcomes determined that uterine congenital AVM should be taken as probable cause of heavy uterine bleeding when refractory intrauterine bleeding is presented. Colour Doppler sonography should be employed for assessment of lesions size and sites. This has effect of facilitating in determining if the patient requires medical or surgical intervention. Total abdominal hysterectomy should be used as the best option when a life threatening haemorrhage presents.

The woman case of congenital Bilateral AV malformations helped to provide clinical evidence that large lesions require surgical interventions. The lesions should be associated with subendometrial tissues to qualify for surgical intervention. On the other hand, lesions that are associated with myometrium should be managed through conservative methods.

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References

  1. Elia G.C.: Singer SJ. Uterine artery malformation as a hidden cause of severe uterine bleeding. A case report. J Reprod Med. 2001; 46:389–400.
  2. Flynn MK, Levine D. The noninvasive diagnosis and management of arteriovenous malformation. Obstet Gynaecol 1996; 88:650–2.
  3. Ghosh TK. Arteriovenous malformation of the uterus and pelvis.Obstet Gynaecol. 1986; 68:40–3.
  4. Jain KA, Jeffrey RB Jr, Sommer FG. Gynaecologic vascular abnormailties: diagnosis with Doppler ultrasonography. Radiology 1991; 1:549–51.
  5. Rebarber, A.; Fox, N.S.; Eckstein, D.A.; Lookstein, R.A.; Daniel H. Successful Bilateral Uterine Artery Embolization During an Ongoing Pregnancy, Obstetrics & Gynecology: (2009) 113(2), Part 2 – pp 554-556.
  6. Timmerman D, Van-den B.T, Peeraer K, et al. Vascular malformation in the uterus: ultrasonographic diagnosis and conservative management. Eur J Obstet Gynaecol Reprod Biol. 2000; 92:171–8.
  7. Kwo, J.H.; kin, G.S. Obstetric Iatrogenic Arterial injuries of the uterus: diagnosis with ultrasound and treatment with transcatheter arterial embolization, Radiographics, 2002:22:35-46.

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StudyCorgi. (2022, March 23). A Rare Case of Bilateral Congenital A V Malformation. Retrieved from https://studycorgi.com/a-rare-case-of-bilateral-congenital-a-v-malformation/

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StudyCorgi. "A Rare Case of Bilateral Congenital A V Malformation." March 23, 2022. https://studycorgi.com/a-rare-case-of-bilateral-congenital-a-v-malformation/.

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StudyCorgi. 2022. "A Rare Case of Bilateral Congenital A V Malformation." March 23, 2022. https://studycorgi.com/a-rare-case-of-bilateral-congenital-a-v-malformation/.

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StudyCorgi. (2022) 'A Rare Case of Bilateral Congenital A V Malformation'. 23 March.

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