Angioembolization of Scrotal Arteriovenous Malformations: A Case Report and Literature Review.
Scrotal swelling is a very common condition in medical practice. Differential diagnosis includes benign lesions that affect any component of the scrotum (testicles, soft tissues, and vascular plexus), malignancy, metastases, and infection.
Benign vascular lesions are common in adult males. Varicoceles are the most common lesion, and the rarest is arteriovenous malformations (AVMs). These lesions may present in a broad spectrum of symptoms such as swelling, bleeding, acute pain, sensation of heaviness, and mass-like structure.
AVMs are rare congenital abnormal underdeveloped vascular lesions that grow with the child. They present with a mass-like structure with audible or tangible bruit. The duplex ultrasound study shows high systolic flow, low resistive index, and arterialization of venous waveforms. Magnetic resonance imaging (MRI) helps to define the extent of the AVM and the adjacent tissues. Angiograph shows a rapid transit shunting of contrast agent from feeding arteries to enlarged veins through an underdeveloped vascular plexus .
The most common locations of arteriovenous malformations (AVMs) are intracranial, followed by extracranial head and neck, extremity, and truncal and visceral sites. However, congenital scrotal AVM is a rare condition .
We are reporting a case of huge scrotal AMV managed by angioembolization. We also reviewed data from other similar reported cases in the medical literature.
2. Case Presentation
A 19-year-old male patient presented to our center in September 2017 with progressive diffused swelling in the scrotum. The swelling developed dramatically over the previous year with flashing skin and local warmth. There was no pain, urological symptoms, or bleeding. No medical or trauma histories were reported.
Physical examination revealed an asymmetric diffused scrotal swelling (more progressed in the left side) (Figure 1), local warmth, and scrotal skin flashing. By palpation, there was no pulse or thrill. Only the right testicle was palpable.
Duplex ultrasound (DUS) showed prominent vessels exhibiting both venous and arterial components (Figure 2), with unusual high flow velocities, and dilated veins up to 1 cm in width. The skin thickness was 2 mm at its max.
Computed tomography arteriography showed a large AVM in the scrotum occupying almost all the scrotal sac indenting the testicles that look slightly small, the left testicle was encircled by the malformation. The veins seen were also dilated reaching diameters of 1 cm denoting varicoceles.
The AVM has its arterial supply from the internal iliac arteries and at least one of its veins drained in the left common femoral vein.
Digital subtraction angiography (DSA) demonstrated large AVM in the scrotum being centered mainly on the left side with multiple feeding vessels originating from the anterior division of internal iliac and common femoral arteries bilaterally more apparent from the left sides.
The feeding vessels in the left side were the internal pudendal artery, the superficial external pudendal artery arising from the common femoral artery, and the deep external pudendal artery arising from the deep femoral artery.
In the right side, the internal pudendal artery and a small branch of superficial pudendal artery were noticed.
The conventional scrotal vascular territory is divided into two parts: anterior one-third and posterior two-thirds. The anterior one-third is supplied by the superficial and deep external pudendal arteries, branching from the femoral artery. The posterior two-thirds is supplied by the internal pudendal artery, branching from the internal iliac artery  (Figure 3). The deep external pudendal could be a branch of the deep femoral artery.
Multiple sessions of angioembolization were performed using coils and particles followed by a total resection of the left mass. The patient was informed with all the predictive complications of the procedure.
The first session of embolization was performed in 28/9/2017 in which the left internal pudendal artery and some branches of the superficial external pudendal artery were embolized (Figure 4).
In the second session, one week later, the rest of left superficial external pudendal artery branches were embolized (Figures 5(a)-5(c)). A third session of embolization was needed after two weeks to complete the embolization of the left deep external pudendal artery (Figures 5(d)-5(f)).
Surgical resection was performed in the following day to resect the whole left side of the scrotum, the left testicle was fixed to the right side (Figure 6).
The histopathological study of resected lesions showed vascular vessels with variation in diameters, some hyaline degeneration in the vessels wall and abnormality in arteriovenous structure.
The follow-up during the next 12 months was favorable with good healing of the scrotal incision and no recurrence of lesion (Figure 7).
3. Literature Review
There are many types of vascular lesions that affect the scrotum or the testicle, Sule et al.  divided these into four main types:
(1) Varicoceles: common lesions that affect the spermatic cord. They consist of dilated veins with no bruit
(2) Hemangiomas: rare lesions that affect the scrotum, consist of dilated veins and capillaries with no bruit detected
(3) Lymphangiomas: rare lesions that affect the scrotum, consist of lymphatic ducts with no flow or bruit detected
(4) Arteriovenous malformations: very rare lesions that affect the scrotum, consist of abnormal microfistulas (nidus) between arteries and veins with no capillary bed, high flow, and bruit
Scrotal arteriovenous malformations are very rare. Therefore, there have been no clear recommendations on their treatment.
Until this case, there have been 17 published case reports about scrotal AVMs with variation in clinical presentation and medical management which we summarize in Table 1.
Scrotal AVMs are uncommon lesions in medical practice with many types of treatment approaches. There is no medical study to support one approach over another due to the scarcity of this condition. Although embolization of feeding vessels helps in reducing the risk of massive bleeding during surgical resection in most cases, it also raises the risk of radiation complications including malignancy, sperm DNA mutations, and permanent damage to the testicular tissue.
The most common presentation was swelling or infiltrating mass [3-17], followed by pain [4, 5, 11, 13, 16, 17], and bleeding or ulceration [3-5, 8, 11, 14]. Infertility was the primary presentation in few cases [7, 10].
The main diagnostic method was DUS [3-7, 10-14, 16, 17], as we noticed before, followed by DSA [3-5, 7, 10, 12, 14, 16], especially in patients whom treated with endovascular embolization. MRI was used in some cases to confirm the diagnosis . CTA is a helpful method to diagnose the lesion but not effective for establishing the management ; therefore, DSA is still the golden standard.
Laboratory tests were limited to the patient's condition. Although sperm analysis was commonly abnormal, it was not a routine test, especially in cases of acute pain or bleeding or with patients who refused to do the test (as in our case).
Percutaneous sclerotherapy into the nidus has been described in low-flow vascular malformation treatment. To our best knowledge, the case written by So et al. is the only case report that described an endovascular method to treat a scrotal AVM with transcatheter coil embolization and percutaneous sclerotherapy. In treating the scrotal lesion with direct puncture, which is a very painful procedure, general anesthesia is required to reduce pain and minimize patient movement. In addition, compression during injection helps in localizing the sclerotherapy and reducing the embolic risk .
Embolization materials were variable (coils, gelatin sponge, polyvinyl alcohol sponge, butyl cyanoacrylate mixed with lipodol, particles, and onyx); in our case, we used coils and particles.
Choosing the embolization agent depends on the indication and the experience at hand. Each agent has its specific advantages and disadvantages. Factors influencing the choice of material comprise the size of the target vessel, flow velocity, and duration of embolization (permanent/temporary), see Table 2 .
Skin necrosis was described in some cases with angioembolization treatment. Later, surgical approach was needed to remove the necrotic tissue (as in our case).
Some of the possible complications for embolization procedure  in pelvic area are as follows:
(i) Necrosis of the skin, bladder, or other intraabdominal organs has also been described especially with sever unspecified embolization
(ii) Impotence: bilateral internal pudendal artery embolization
(iii) Arterial perforation
(iv) Hemoglobinuria may develop as a consequence of the hemolysis that follows any sclerosant injection
(v) Allergic reaction due the use of contrast agent or sclerotherapy agents
(vi) Acute kidney injury due the contrast agent
(vii) Local complications in the insertion artery (hematoma, pseudoaneurysm)
(viii) Delayed malignancy due to the exposure to high dosage of radiation
(ix) DNA mutations of sperm cells/ovarian cells or infertility due to radiation
Conservative approach with periodic assessment was an option in some cases especially in young asymptomatic patients.
The scarcity of scrotal AVMs and the variety of medical approaches present a challenge in both diagnostic and treatment approaches.
We should think of AVMs in patients with rapidly developed lesions despite other symptoms such as pain or bleeding, without neglecting other deferential diagnoses, mainly tumors and infections.
Doppler US is very helpful for diagnosis of these lesion but is not sufficient for planning the management. Patients should be fully informed about the complications of each treatment plan in order to give an informed consent.
The management should consider the individuality of each case.
We could not do a sperm count (because the patient refused). There was no intervention in the right side of the scrotum due to the hesitation of the patient.
The patient gave written informed consent to the publication of his case details and clinical photographs.
Conflicts of Interest
The authors declare that there are no conflicts of interest regarding the publication of this paper.
The authors thank Dr. Loay Alia, Dr. Fadi Kasoha, and Dr. Montazar Bazal from of Al-Assad University Hospital for their contribution in the surgical intervention. Also, they would like to thank Dr. Omar Tillo for linguistic revision.
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Ammar Mohammad, (1) Wael Sahyouni, (1) Taisser Almeree, (2) and Bayan Alsaid [ID] (3,4)
(1) Department of Vascular Surgery, Al-Assad University Hospital, Damascus, Syria
(2) Department of Radiology, Al-Assad University Hospital, Damascus, Syria
(3) Department of General Surgery, Al-Assad University Hospital, Damascus, Syria
(4) Laboratory of Anatomy, Faculty of Medicine, University of Damascus, Damascus, Syria
Correspondence should be addressed to Bayan Alsaid; email@example.com
Received 12 September 2019; Accepted 31 January 2020; Published 8 February 2020
Academic Editor: Muzaffer Sindel
Caption: Figure 1: Left scrotal swelling.
Caption: Figure 2: (a, b) Doppler ultrasound showed dilated vessels with both venous and arterial components; (c) computed tomography arteriography.
Caption: Figure 3: Normal blood supply of scrotum.
Caption: Figure 4: Embolization of multiple vessels: (a, b) internal pudendal artery and (c, d, e) superficial external pudendal artery. (f) View of scrotum after 24 hours.
Caption: Figure 5: (a, b) Second session of angioembolization of the rest of superficial external pudendal artery branches; (c) affected area after 24 hours; (d) anterioposterior view of left deep external pudendal artery during the third session. (e) Final view showing no more visible feeding branches to the arteriovenous malformation. (f) Final clinically infarcted area before surgery.
Caption: Figure 6: Surgical steps: (a) isolation of left spermatic cord with the testicle from inguinal incision, (b, c) resection of the lesion, and (d) insertion of the left testicle in the right side of scrotum. (e) View after surgery.
Caption: Figure 7: After 3 months (a). After 12 months (b).
Table 1: Published cases till 1/1/2019. Author Age Thrill/ (year) (years) Presentation bruit Bezirdjian et al. 24 Painless + (1989)  enlarging right scrotal mass Hamid et al. 55 Right scrotal + (1992)  swelling, pain with ulceration and bleeding Sule et al. 17 Intermittently + (1993)  bleeding pulsatile left scrotal mass Konus et al. 8 Progressively + (1999)  enlarging, intermittently bleeding, painful pulsatile scrotal mass Kang et al. 20 Acute scrotal -- (2004)  swelling detected 4 days after a trauma Gonzalez et al. 31 Left scrotal Not (2002)  swelling mentioned with virtual azoospermia. Bandi et al. 67 Recurrent scrotal Not (2004)  AVM-bleeding mentioned nonhealing ulcer 12 years after preoperative embolization and hemiscrotectomy Monoski et al. 31 Primary infertility -- (2006)  and left scrotal fullness Yilmaz et al. 51 Pain and Pulsatile (2009)  throbbing vessels + sensation in right hemiscrotum Jaganathan et al. 2 2 cases both Not (2011)  32 presented mentioned with scrotal swelling and bleeding Zachariah et al. 30 Progressive -- (2012)  swelling. One episode of acute pain before 4 mo. Key R. et al. 41 Massive bleeding -- (2013)  after a hip fracture due to trauma. With large right retro peritoneal hematoma Sato et al. 38 Recurrent scrotal -- (2013)  mass So WL et al. 26 Scrotal pain and -- (2014)  swelling Muslim et al. 16 Right scrotal Not (2014)  swelling mentioned associated with mild pain Our case 19 Left scrotum + swelling Author Sperm (year) analysis Investigation Bezirdjian et al. Not done US. (1989)  Arteriogram Hamid et al. Azoospermia Low (1992)  s.testosterone, DUS, DSA Sule et al. -- DSA (1993)  Konus et al. Not done DUS, DSA (1999)  Kang et al. Not done DUS (2004)  Gonzalez et al. Azoospermia DUS, DSA (2002)  Bandi et al. Not done Not done at (2004)  second presentation Monoski et al. Severe DSA (2006)  oligospermia hypertrophied internal pudendal and branch of superficial femoral a. Yilmaz et al. Not done Scrotal (2009)  ultrasound. Confirmed at DUS Jaganathan et al. Not done DUS, DSA (2011)  Emergency DUS, DSA Zachariah et al. Not done DUS, MRI (2012)  Key R. et al. -- DUS, DSA (2013)  Sato et al. -- CTA, biopsy (2013)  (micro-AV fistula--AVM) So WL et al. Not DUS (AVM). (2014)  mentioned DSA Muslim et al. Not DUS (2014)  mentioned Our case Not DUS, MCT, performed DSA Author (year) Management Follow-up Bezirdjian et al. Angioembolization Not (1989)  (polyvinyl alcohol mentioned sponge (Ivalon)). Surgical debridement Hamid et al. Angioembolization Not (1992)  then surgery commented Sule et al. Angioembolization No recurrence (1993)  (gelatin sponge at 2 years of and coils) follow-up failed. Complete surgical resection was done Konus et al. Angioembolization 1 year later. No (1999)  (polyvinyl alcohol residual sponge). Surgical disease on excision follow-up Doppler Kang et al. Surgical Not (2004)  excision. Biopsy mentioned showed AVM Gonzalez et al. Bilateral 3 months of (2002)  varicocelectomy. follow-up, Super selective sperm analyses angioembolization improved followed by surgical excision Bandi et al. Surgical excision. Not (2004)  mentioned Monoski et al. Bilateral Sperm count (2006)  varicocelectomy. improved. 3 Angioembolization. years later, Surgical excision successful spontaneous pregnancy Yilmaz et al. Not mentioned Not (2009)  mentioned Jaganathan et al. Selective 13 months of (2011)  angioembolization follow-up, (poly vinyl alcohol). asymptomatic. Parents refused surgery. 18 months of Angioembolization follow-up, no (n-butyl cyanoacrylate recurrence mixed with lipodol) Zachariah et al. Angioembolization Not (2012)  was rejected. mentioned Surgical excision. Key R. et al. Several sessions of 18 weeks later, (2013)  angioembolization no symptoms (micro coils/emposphere's/gel foam particles/onyx Sato et al. Surgical excision 10 months (2013)  later, no recurrence So WL et al. Coil embolization. 3 months of (2014)  Subcutaneous review, no sclerosant recurrence (sodium tetradecyl sulfate 3% with ethiodised oil (2 : 1 ratio) Muslim et al. Refused embolization. One-year (2014)  Surgical excision with follow-up, no dissecting the spermatic recurrence cord through an inguinal incision to protect it Our case Three sessions of One-year embolization, follow-up, no resection of recurrence scrotal lesion Table 2: Types of embolization agents. Embolization agents Organic Autologous clots, fibrin Coils Standard coils, detachable coils, active coils Plugs, balloons Detachable balloon, vascular plug Alcohol, polymerizing substances (histoacryl), Liquids, detergents (e.g., fibrovein, ethoxysclerol), sclerosants antibiotics (e.g., doxocycline, bleomycin), precipitating substances (onyx[R]) Particles Gelfoam, polyvinyl alcohol particles, spherical particles (e.g., spherical PVA, acryl polymere)
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|Title Annotation:||Case Report|
|Author:||Mohammad, Ammar; Sahyouni, Wael; Almeree, Taisser; Alsaid, Bayan|
|Publication:||Case Reports in Vascular Medicine|
|Date:||Mar 1, 2020|
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