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CASE REPORT
Year : 2022  |  Volume : 25  |  Issue : 7  |  Page : 1189-1191

A postpartum arteriovenous malformation case diagnosed with late postpartum bleeding


1 Department of Obstetrics-Gynecology, Etlik Zübeyde Hanım Maternity and Women's Health Teaching and Research Hospital Ankara, Turkey
2 Department of Perinatology, Etlik Zübeyde Hanım Maternity and Women's Health Teaching and Research Hospital Ankara, Turkey

Date of Submission13-Oct-2021
Date of Acceptance19-Mar-2022
Date of Web Publication20-Jul-2022

Correspondence Address:
Dr. E N Varli
Department of Obstetrics-Gynecology, Etlik Zübeyde Hanim Maternity and Women's Health Teaching and Research Hospital, Ankara
Turkey
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/njcp.njcp_1883_21

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   Abstract 


Postpartum hemorrhage is the most important and also preventable cause of maternal morbidity and mortality worldwide. Arteriovenous malformation (AVM) is a rare cause of postpartum hemorrhage. We present a case of arteriovenous malformation, which may be one of the rare but potentially fatal causes of postpartum hemorrhage (PPH). In the case report, on the postpartum 52nd day, the patient presented to the emergency department with a complaint of vaginal bleeding. Doppler ultrasonography revealed a lesion compatible with the vascular structure, the largest diameter of 28 * 28 mm, in the uterus. The patient was found not eligible for embolization, and a hysterectomy was then indicated. After the hysterectomy, the pathology diagnosis supported the initial clinical and radiological suspicions of AVM. Hysterectomy is the definitive treatment method of AVM and should be considered in patients who are not eligible for embolization. This case is presented to draw the attention of physicians to AVM as a possible cause of secondary PPH.

Keywords: Arteriovenous malformations, Doppler ultrasonography, hysterectomy, postpartum hemorrhage, uterine artery embolization


How to cite this article:
Ozturk A C, Varli E N, Caglar A T, Korkmaz V. A postpartum arteriovenous malformation case diagnosed with late postpartum bleeding. Niger J Clin Pract 2022;25:1189-91

How to cite this URL:
Ozturk A C, Varli E N, Caglar A T, Korkmaz V. A postpartum arteriovenous malformation case diagnosed with late postpartum bleeding. Niger J Clin Pract [serial online] 2022 [cited 2022 Oct 3];25:1189-91. Available from: https://www.njcponline.com/text.asp?2022/25/7/1189/351462




   Introduction Top


Postpartum hemorrhage is the most important and preventable cause of maternal morbidity and mortality worldwide. The bleeding between 24 hours and 12 weeks after delivery is defined as secondary or late postpartum hemorrhage.[1] The most common causes of late postpartum bleeding are retention of conception products, endometritis, incomplete involution of the placental implantation site, and coagulation disorders. Uterine artery pseudoaneurysm, choriocarcinoma, and arteriovenous malformation (AVM) are other rare causes.[2] AVMs can be classified as acquired and congenital, whereas congenital forms are caused by embryological differentiation failure leading to abnormal vascular connections. Acquired AVMs are detected after uterine surgeries such as cesarean, dilatation curettage, and myomectomy that erode endometrial vessels.[3]

The AVM can be diagnosed by Doppler ultrasonography, magnetic resonance imaging (MRI), and computed tomography (CT) angiography. Radiographic angiography remains the gold standard.[4] In histological evaluation, intertwined vascular structures are observed in the myometrium.[5] The most effective and safe method of treatment is uterine artery embolization. A hysterectomy is also an option if the patient does not want to preserve fertility, wants definitive treatment, or other methods are not suitable.[6]

By sharing this case, we aimed to draw attention to arteriovenous malformations, which may be one of the rare but potentially fatal causes of postpartum hemorrhage.


   Case Report Top


The patient was 34 years old when she applied to the emergency department and had a history of seven gravidas and three paras. She had one previous cesarean (C/S). Her complaints were groin pain contractions. Our patient, who was 28 weeks pregnant, had not previously undergone pregnancy follow-up. Increased vascularity showing intense arterial and venous turbulent flow was observed in the subchorionic area extending from the uterus fundus to the cervix in the ultrasonography.

The patient was hospitalized because of a high-risk pregnancy. The emergency cesarean was performed due to heavy vaginal bleeding in the 30th week while hospitalized for 2 weeks. A 1785-g live baby girl was delivered with APGAR 6/8. When intraoperative bleeding occurred in the lower segment, a Bakri balloon was placed, and the surgery was completed by performing a tubal ligation upon the patient's request. A Bakri balloon was removed at the 24th hour in the postoperative follow-up. The patient, who did not have any problems, was discharged on the 6th postoperative day. The patient who could have spontaneous resolution was followed up.

However, on the postpartum 52nd day, the patient presented to the emergency department with a complaint of vaginal bleeding. In the ultrasonography performed, the uterus was observed with 137 * 70 mm larger than normal. Uterine involution was insufficient. Doppler ultrasonography revealed that a lesion compatible with the vascular structure, the largest of 28 * 28 mm, was observed in the uterus [Figure 1]. In the laboratory findings, Hb: 9.1 gr/dl, htc: 27.3, plt: 257.000. There was extensive active vaginal bleeding. Due to that, embolization was not applied to the patient consulted with the interventional radiology department. In this case of arteriovenous malformation, a hysterectomy was planned with heavy bleeding and no fertility intention. Due to the size of the uterus, a midline incision was preferred for entry into the abdomen. The uterine lower segment was larger than expected, and vascularization increased [Figure 2] and [Figure 3].
Figure 1: Preoperative USG view

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Figure 2: Intraoperative view of arteriovenous malformation

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Figure 3: Intraoperative view of the uterine cavity

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The bladder was adhered to the lower uterine segment due to previous operations. After the hysterectomy was completed, the defect in the bladder was repaired by double-layer continuous suture with 3/0 vicryl. Then the operation was completed by placing a soft drain in the abdomen. In the postoperative follow-up, the patient's drain was removed on the 2nd postoperative day, and the patient was discharged on the 3rd day. The urinary catheter of the patient was removed on the 10th postoperative day. Pathology results reported scattered vascular structures of varying sizes, some intertwined, some with dilated lumen, most thick-walled, which do not form a limited mass in the myometrium.


   Discussion Top


AVM is a rare entity, which may be due to under-reporting. Uterine AVM can be symptomatic or asymptomatic.[7] Six percent of asymptomatic AVMs resolved spontaneously. Conservative management is effective in two-thirds of patients diagnosed with AVM sonographically.[3] We discharged our patient on the 6th day with the expectation of spontaneous resolution. However, the spontaneous resolution did not occur in our patient. Our patient, who had no fertility intention, was admitted to the emergency service with heavy bleeding on the postpartum 52nd day.

As in our case, hypervascular, turbulent tortuous vessels can be monitored with Doppler ultrasonography. It should be kept in mind in differential diagnosis.[7] Although color Doppler ultrasound is currently the most widely used method, the gold standard for diagnosing arteriovenous malformation is angiography.[4] Angiography is advantageous because it can be treated simultaneously at diagnosis.

Symptomatic AVMs require intervention in the form of arterial embolization, uterine artery ligation, or hysterectomy.[8] In a systematic review of 100 patients, it was reported that uterine artery embolization (UAE) was the most common treatment option (59%), followed by hysterectomy (29%).[3]

UAE is safe, fertility can be preserved, but hemodynamics are unstable, hysterectomy is the definitive treatment method in patients who are not suitable for postmenopausal or embolization.[5] In our case, a hysterectomy was preferred because the patient was not suitable for embolization. Also in other cases in the literature, hysterectomy was chosen in patients with heavy bleeding who are not suitable for embolization. There are also cases of arteriovenous malformation associated with placenta accreta, which is the cause of late postpartum bleeding. Hysterectomy was also performed in these cases.[9]

Color Doppler ultrasonography should always be performed to diagnose unexplained uterine bleeding. Vascular lesions in the uterus are more common than we might have guessed, and their prevalence is increasing with increasing cesarean-curettage rates.

Patients who apply with late postpartum bleeding arteriovenous malformation should also be considered in the differential diagnosis. The patient's vitals, fertility request, and embolization eligibility criteria should be considered in their treatment.

Abbreviations

AVM = Arteriovenous malformation

MRI = Magnetic resonance imaging

CT = Computed tomography

C/S = Cesarean

IUGR = Intrauterine growth restriction

UAE: Uterine artery embolization

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Hoveyda F, MacKenzie I. Secondary postpartum haemorrhage: Incidence, morbidity and current management. Br J Obstetr Gynaecol 2001;108:927-30.  Back to cited text no. 1
    
2.
Oyelese Y, Ananth CV. Postpartum hemorrhage: Epidemiology, risk factors, and causes. Clin Obstetr Gynecol 2010;53:147-56.  Back to cited text no. 2
    
3.
Peitsidis P, Manolakos E, Tsekoura V, Kreienberg R, Schwentner L. Uterine arteriovenous malformations induced after diagnostic curettage: A systematic review. Arch Gynecol Obstetr 2011;284:1137-51.  Back to cited text no. 3
    
4.
Eling R, Kent A, Robertson M. Pregnancy after uterine arteriovenous malformation–case series and literature review. Australas J Ultrasound Med 2012;15:87-96.  Back to cited text no. 4
    
5.
Szpera-Goździewicz A, Gruca-Stryjak K, Bręborowicz GH, Ropacka-Lesiak M. Uterine arteriovenous malformation-diagnosis and management. Ginekol Pol 2018;89:276-9.  Back to cited text no. 5
    
6.
Letailleur M, Dietrich G, Marpeau L, Roman H. Hysterectomy for uterine arteriovenous malformation: Laparoscopic view. J Minim Invasive Gynecol 2016;23:158-9.  Back to cited text no. 6
    
7.
Timmerman D, Van den Bosch T, Peeraer K, Debrouwere E, Van Schoubroeck D, Stockx L, et al. Vascular malformations in the uterus: Ultrasonographic diagnosis and conservative management. Eur J Obstetr Gynecol Reprod Biol 2000;92:171-8.  Back to cited text no. 7
    
8.
Ogelzang RL, Nemcek AA Jr, Skrtic Z, Gorrell J, Lurain JR. Uterine arteriovenous malformations: Primary treatment with therapeutic embolization. J Vasc Interv Radiol 1991;2:517-22.  Back to cited text no. 8
    
9.
Lalitha N, Seetha P, Shanmugasundaram R, Rajendiran G. Uterine arteriovenous malformation: Case series and literature review. J Obstet Gynaecol India 2016;66:282-6.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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