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Angioembolization of Scrotal Arteriovenous Malformations: A Case Report and Literature Review.

1. Introduction

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 [1].

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 [2].

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 [3] (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. [4] 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.

4. Discussion

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 [13]. CTA is a helpful method to diagnose the lesion but not effective for establishing the management [15]; 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 [3].

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 [19].

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 [20] 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.

5. Conclusion

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.

6. Limitations

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.

https://doi.org/10.1155/2020/8373816

Consent

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.

Acknowledgments

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.

References

[1] K. Hubert, J. S. Elder, and G. T. MacLennan, "Benign vascular lesions of the scrotum," The Journal of Urology, vol. 176, no. 5, p. 2245, 2006.

[2] J. J. Marler and J. B. Mulliken, "Current management of hemangiomas and vascular malformations," Clinics in Plastic Surgery, vol. 32, no. 1, pp. 99-116, ix, 2005.

[3] W. L. A. So, J. Chaganti, R. Waugh, and R. J. Ferguson, "Management of scrotal arteriovenous malformation with transcatheter embolisation coils and percutaneous sclerotherapy under angiographic guidance," Journal of Medical Imaging and Radiation Oncology, vol. 59, no. 4, pp. 468-470, 2015.

[4] J. D. Sule, M. J. Lemmers, and J. M. Barry, "Scrotal arteriovenous malformation: case report and literature review," The Journal of Urology, vol. 150, no. 6, pp. 1917-1919, 1993.

[5] D. R. Bezirdjian, P. D. Reznikov, and J. Tisnado, "Transcatheter embolization of an arteriovenous malformation of the scrotum," Cardiovascular and Interventional Radiology, vol. 12, no. 5, pp. 267-269, 1989.

[6] S. Hamid, J. Aquilina, W. Davidson, and C. Dhabuwala, "Arteriovenous malformation of scrotum: a case report," The Journal of Urology, vol. 147, no. 1, pp. 160-162, 1992.

[7] O. Konus, E. Ilgit, C. Yucel, E. Ozbek, and B. Onal, "Scrotal arteriovenous malformation and its preoperative embolization," European Radiology, vol. 9, no. 3, pp. 425-427, 1999.

[8] T. W. Kang, Y.-D. Choi, Y. Y. Jeong et al., "Intrascrotal extratesticular arteriovenous malformation," Urology, vol. 64, no. 3, p. 590, 2004.

[9] R. R. Gonzalez, A. J. Thomas Jr., and M. Goldstein, "Arteriovenous malformation of the scrotum causing severe oligospermia," Fertility and Sterility, vol. 78, article S210, 2002.

[10] G. Bandi, F. J. Bianco, and C. Dhabuwala, "Recurrent scrotal arteriovenous malformation," Journal of Urology, vol. 171, no. 4, p. 1628, 2004.

[11] S. H. Choi, J. H. Lee, D. J. Kim, and S. R. Cho, "Scrotal arteriovenous malformation," Korean Journal of Urology, vol. 46, no. 8, pp. 873-875, 2005.

[12] M. A. Monoski, R. R. Gonzalez, A. J. Thomas, and M. Goldstein, "Arteriovenous malformation of scrotum causing virtual azoospermia," Urology, vol. 68, no. 1, pp. 203.e5-203.e6, 2006.

[13] C. Yilmaz, M. Arslan, and M. Arslan, "Intrascrotal arteriovenous malformation simulating varicocele," American Journal of Roentgenology, vol. 192, no. 6, article W351, 2009.

[14] S. Jaganathan, S. Gamanagatti, A. Mukund, and A. Dhar, "Bleeding scrotal vascular lesions: interventional management with transcatheter embolization," CardioVascular and Interventional Radiology, vol. 34, no. S2, pp. 113-116, 2011.

[15] J. R. Zachariah, A. K. Gupta, and S. Lamba, "Arteriovenous malformation of the scrotum: is preoperative angioembolization a necessity?," Indian Journal of Urology, vol. 28, no. 3, pp. 329-334, 2012.

[16] R. Key and T. Bishop, "Successful use of onyx[R] for transcatheter arterial embolization of a scrotal arteriovenous malformation refractory to traditional embolic agents," Radiology Case Reports, vol. 8, no. 3, p. 846, 2013.

[17] N. Sato, T. Kuroki, Y. Tosa, T. Kusano, and S. Yoshimoto, "A scrotal arteriovenous malformation: a case report," Modern Plastic Surgery, vol. 4, no. 1, pp. 16-19, 2014.

[18] S. Muslim, N. Roy, and I. Alam, "Scrotal arteriovenous malformation excision-an inguinal approach," Journal of Clinical Urology, vol. 10, no. 3, pp. 227-229, 2017.

[19] M. Smits, "Embolization materials and tecnique 2017," January 2020, https://angiofellow.com/author/mrtnsmits/last.

[20] J. I. Bilbao, A. Martinez-Cuesta, F. Urtasun, and O. Cosin, "Complications of embolization," Seminars in Interventional Radiology, vol. 23, no. 2, pp. 126-142, 2006.

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; drbayan@gmail.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) [5]                          enlarging right
                                      scrotal mass

Hamid et al.             55          Right scrotal            +
(1992) [6]                             swelling,
                                       pain with
                                       ulceration
                                      and bleeding

Sule et al.              17         Intermittently            +
(1993) [4]                              bleeding
                                       pulsatile
                                      left scrotal
                                          mass

Konus et al.             8           Progressively            +
(1999) [7]                             enlarging,
                                    intermittently
                                       bleeding,
                                   painful pulsatile
                                      scrotal mass

Kang et al.              20          Acute scrotal            --
(2004) [8]                              swelling
                                    detected 4 days
                                     after a trauma

Gonzalez et al.          31          Left scrotal            Not
(2002) [9]                              swelling          mentioned
                                      with virtual
                                      azoospermia.

Bandi et al.             67        Recurrent scrotal         Not
(2004) [10]                           AVM-bleeding        mentioned
                                   nonhealing ulcer
                                    12 years after
                                      preoperative
                                    embolization and
                                    hemiscrotectomy

Monoski et al.           31       Primary infertility         --
(2006) [12]                            and left
                                    scrotal fullness

Yilmaz et al.            51            Pain and           Pulsatile
(2009) [13]                            throbbing          vessels +
                                     sensation in
                                         right
                                      hemiscrotum

Jaganathan et al.        2           2 cases both            Not
(2011) [14]              32            presented          mentioned
                                     with scrotal
                                        swelling
                                      and bleeding

Zachariah et al.         30           Progressive             --
(2012) [15]                            swelling.
                                    One episode of
                                      acute pain
                                      before 4 mo.

Key R. et al.            41        Massive bleeding           --
(2013) [16]                           after a hip
                                    fracture due to
                                   trauma. With large
                                      right retro
                                      peritoneal
                                        hematoma

Sato et al.              38        Recurrent scrotal          --
(2013) [17]                               mass

So WL et al.             26         Scrotal pain and          --
(2014) [3]                              swelling

Muslim et al.            16          Right scrotal           Not
(2014) [18]                            swelling           mentioned
                                    associated with
                                       mild pain

Our case                 19          Left scrotum             +
                                        swelling

Author                    Sperm
(year)                   analysis       Investigation

Bezirdjian et al.        Not done            US.
(1989) [5]                               Arteriogram

Hamid et al.           Azoospermia           Low
(1992) [6]                             s.testosterone,
                                           DUS, DSA

Sule et al.                 --               DSA
(1993) [4]

Konus et al.             Not done          DUS, DSA
(1999) [7]

Kang et al.              Not done            DUS
(2004) [8]

Gonzalez et al.        Azoospermia         DUS, DSA
(2002) [9]

Bandi et al.             Not done        Not done at
(2004) [10]                                 second
                                         presentation

Monoski et al.            Severe             DSA
(2006) [12]            oligospermia     hypertrophied
                                           internal
                                         pudendal and
                                          branch of
                                         superficial
                                          femoral a.

Yilmaz et al.            Not done          Scrotal
(2009) [13]                              ultrasound.
                                         Confirmed at
                                             DUS

Jaganathan et al.        Not done          DUS, DSA
(2011) [14]                               Emergency
                                           DUS, DSA

Zachariah et al.         Not done          DUS, MRI
(2012) [15]

Key R. et al.               --             DUS, DSA
(2013) [16]

Sato et al.                 --           CTA, biopsy
(2013) [17]                               (micro-AV
                                        fistula--AVM)

So WL et al.               Not            DUS (AVM).
(2014) [3]              mentioned            DSA

Muslim et al.              Not               DUS
(2014) [18]             mentioned

Our case                   Not            DUS, MCT,
                        performed            DSA

Author
(year)                        Management               Follow-up

Bezirdjian et al.         Angioembolization               Not
(1989) [5]                (polyvinyl alcohol           mentioned
                          sponge (Ivalon)).
                         Surgical debridement

Hamid et al.              Angioembolization               Not
(1992) [6]                   then surgery              commented

Sule et al.               Angioembolization          No recurrence
(1993) [4]                 (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) [7]                (polyvinyl alcohol           residual
                          sponge). Surgical           disease on
                               excision                follow-up
                                                        Doppler

Kang et al.                   Surgical                    Not
(2004) [8]                 excision. Biopsy            mentioned
                              showed AVM

Gonzalez et al.               Bilateral               3 months of
(2002) [9]                varicocelectomy.            follow-up,
                           Super selective          sperm analyses
                          angioembolization            improved
                         followed by surgical
                               excision

Bandi et al.              Surgical excision.              Not
(2004) [10]                                            mentioned

Monoski et al.                Bilateral               Sperm count
(2006) [12]                varicocelectomy.           improved. 3
                          Angioembolization.         years later,
                          Surgical excision           successful
                                                      spontaneous
                                                       pregnancy

Yilmaz et al.               Not mentioned                 Not
(2009) [13]                                            mentioned

Jaganathan et al.             Selective              13 months of
(2011) [14]               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) [15]                 was rejected.              mentioned
                               Surgical
                              excision.

Key R. et al.            Several sessions of        18 weeks later,
(2013) [16]               angioembolization           no symptoms
                                (micro
                        coils/emposphere's/gel
                         foam particles/onyx

Sato et al.               Surgical excision            10 months
(2013) [17]                                            later, no
                                                      recurrence

So WL et al.              Coil embolization.          3 months of
(2014) [3]                  Subcutaneous              review, no
                              sclerosant              recurrence
                          (sodium tetradecyl
                           sulfate 3% with
                           ethiodised oil
                            (2 : 1 ratio)

Muslim et al.           Refused embolization.          One-year
(2014) [18]             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
Geographic Code:7SYRI
Date:Mar 1, 2020
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