Capillary hemangioma of the testis. A case report of a rare benign tumor

Publications

Share / Export Citation / Email / Print / Text size:

Journal of Ultrasonography

Polish Ultrasound Society (Polskie Towarzystwo Ultrasonograficzne)

Subject: Medicine

GET ALERTS

ISSN: 2084-8404
eISSN: 2451-070X

DESCRIPTION

0
Reader(s)
0
Visit(s)

Comment(s)
0
Share(s)

SEARCH WITHIN CONTENT

FIND ARTICLE

Volume / Issue / page

Related articles

VOLUME 16 , ISSUE 64 (March 2016) > List of articles

Capillary hemangioma of the testis. A case report of a rare benign tumor

Waldemar Białek * / Sławomir Rudzki / Lech Wronecki

Keywords : capillary hemangioma of the testis, testicular tumor, three-dimensional ultrasound of the scrotum, orchidectomy

Citation Information : Journal of Ultrasonography. Volume 16, Issue 64, Pages 87-93, DOI: https://doi.org/10.15557/JoU.2016.0009

License : (CC BY-NC-ND 3.0)

Received Date : 16-July-2015 / Accepted: 04-October-2015 / Published Online: 29-March-2016

ARTICLE

ABSTRACT

This paper presents the case of a very rare capillary hemangioma of the testis in a 23-year-old patient. Physical examination revealed a tumor located in the upper pole of the left testis, which was suspected of being malignant due to its significantly increased density and irregular contours. Blood levels of the following tumor markers were determined: alpha-fetoprotein, human chorionic gonadotropin and lactate dehydrogenase. No abnormalities were found in any of these tests. A gray-scale ultrasound scan of the scrotum revealed a lesion located in the upper pole of the left testis, 24 mm in diameter with slightly decreased echogenicity and irregular contours, which suggested infiltration of the tunica albuginea of the testis. Color and power Doppler scans demonstrated a dense network of blood vessels and increased blood flow in the lesion described. In addition, 3D ultrasound scan images were obtained, which allowed for a thorough determination of the topography of the lesion. No abnormalities were found in the patient's medical history, physical examination or ultrasound scan of the right testis. Taking into account the suspected malignancy with signs of infiltration of the tunica albuginea of the upper pole of the testis a decision was made to remove the left testis together with the spermatic cord using the inguinal approach. A histopathological examination of the whole specimen revealed a multifocal capillary hemangioma of the testis, signs of testicular fibrosis and significant atrophy of the spermatogenic epithelium of the seminiferous tubules. Immunohistochemistry: CD31 (+), CD34 (+), FVIII (−), vimentin (+), CK MNF116 (−), mesothelial cells (−), calretinin (−), MIB-1 = 8.4%. The tumor described is one of the few benign lesions originating from the tissues of the testis for which partial resection of the organ may be considered. Intraoperative histopathological examination and resection of the lesion with the preservation of a healthy tissue margin may be considered in patients with a single testis in whom the tumor is found to have a very rich vascular network and the levels of tumor markers are normal.

W niniejszej pracy przedstawiono przypadek bardzo rzadko występującego naczyniaka krwionośnego włośniczkowego jądra u 23-letniego pacjenta. Guz zlokalizowany w badaniu fizykalnym w górnym biegunie jądra lewego ze względu na znacznie wzmożoną spoistość i nieregularne obrysy budził podejrzenie nowotworu złośliwego. Oznaczono stężenia markerów nowotworowych we krwi: alfa-fetoproteiny, gonadotropiny kosmówkowej oraz dehydrogenazy mleczanowej, nie stwierdzając odchyleń od normy w żadnym z tych badań. W badaniu ultrasonograficznym moszny wykonanym w skali szarości w górnym biegunie jądra lewego zaobserwowano ognisko o średnicy 24 mm o nieznacznie obniżonej echogeniczności i nieregularnych obrysach, sugerujących naciekanie błony białawej jądra. W opcji dopplera kolorowego i dopplera mocy stwierdzono gęstą sieć naczyń krwionośnych oraz wzmożone przepływy krwi w opisywanej zmianie. Dokonano również akwizycji obrazów w ultrasonograficznym badaniu trójwymiarowym, które pozwoliło na poznanie dokładnej topografii ogniska. W badaniu podmiotowym, przedmiotowym oraz ultrasonograficznej ocenie jądra prawego nie stwierdzono nieprawidłowości. Biorąc pod uwagę podejrzenie nowotworu złośliwego, o cechach naciekania błony białawej górnego bieguna jądra, pacjenta zakwalifikowano do usunięcia jądra lewego wraz z powrózkiem nasiennym z dostępu przez kanał pachwinowy. W badaniu histopatologicznym całego preparatu stwierdzono wieloogniskowego naczyniaka krwionośnego włośniczkowego jądra, cechy włóknienia jądra oraz znacznego stopnia atrofię nabłonka plemnikotwórczego kanalików nasiennych. Immunohistochemicznie: CD31 (+), CD34 (+), FVIII (−), wimentyna (+), CK MNF116 (−), komórki mezotelialne (−), kalretynina (−), MIB-1 = 8,4%. Opisywany nowotwór jest jedną z nielicznych niezłośliwych zmian wywodzących się z tkanek jądra, w przypadku których można rozważać resekcję częściową narządu. U pacjentów z jądrem jedynym, u których stwierdza się guz o bardzo bogatej sieci naczyń, przy prawidłowych stężeniach markerów nowotworowych można rozważać śródoperacyjne badanie histopatologiczne i resekcję zmiany z zachowaniem marginesu zdrowych tkanek.

Graphical ABSTRACT

Case report

A 23-year-old man reported to a urologist due to a tumor of the left testis. Two months earlier he had noticed that the upper part of the testicle was characterized by increased density and irregular contours. The patient denied trauma to the scrotum; in addition, no symptoms which could suggest inflammation of the epididymis or the left testis were found. The man only reported a slight discomfort in this area.

Physical examination revealed a hard tumor not painful on palpation, 2 cm in diameter, with uneven contours, located in the upper pole of the left testis. No abnormalities were observed in the spermatic cords or the right epididymis and testis. Blood lactate dehydrogenase (LDH), alpha-fetoprotein (AFP) and human chorionic gonadotropin (beta-hCG) assays were recommended. No abnormalities were demonstrated in any of these tests.

An ultrasound scan of the scrotum was performed. An oval lesion 24 mm in diameter with slightly decreased echogenicity was visualized in the upper pole of the left testis (Fig. 1). No calcifications, fibrosis or foci of necrotic tissue were found in the area of the lesion. The left testis was surrounded by approximately 2 ml of clear fluid. The lesion seemed to infiltrate and cross the tunica albuginea next to the head of the epididymis (Fig. 2).

Fig. 1

Ultrasound scan. 12 MHz linear probe. Left testis. A lesion with a slightly decreased echogenicity is visible in its upper pole

JoU-2016-0009-g001.jpg
Fig. 2

Ultrasound scan. 12 MHz linear probe. Irregular contours of the upper pole of the left testis which suggest infiltration of the tunica albuginea

JoU-2016-0009-g002.jpg

The tumor was characterized by very rich vasculature and very distinct blood flow in color and power Doppler scans (Fig. 3 and 4). The arrangement of the vessels resembled that of a normal vasculature pattern of the testicular parenchyma. 3D ultrasound images were obtained using the free-hand technique and color Doppler option (Fig. 5) and a three-dimensional reconstruction of the suspected lesion vasculature was performed (Fig. 6). No other abnormalities were found in the ultrasound scan of the scrotum.

Fig. 3

Ultrasound scan with color Doppler imaging. The vasculature of the upper pole of the left testis is distinctly larger than that of the remaining testicular tissue

JoU-2016-0009-g003.jpg
Fig. 4

Ultrasound scan with power Doppler imaging. The examination confirms a significantly larger blood flow in the upper pole of the testis in the area of the previously found lesion

JoU-2016-0009-g004.jpg
Fig. 5

3D ultrasound scan. Three-dimensional reconstruction of the left testis using color Doppler imaging

JoU-2016-0009-g005.jpg
Fig. 6

3D ultrasound scan. Three-dimensional reconstruction of the vasculature of the suspicious lesion located in the upper pole of the left testis allows for the determination of its precise topography

JoU-2016-0009-g006.jpg

A decision was made to remove the left testis. A left inguinal incision was made to reach the spermatic cord and isolate the left testis together with the tunica vaginalis from the scrotum. The testis could be freely moved inside the tunica vaginalis. Macroscopically the upper pole of the testis was notable for its bluish color; it was hard on palpation with uneven surface focally (Fig. 7). Due to the suspected malignant tumor originating from the upper pole of the testis the organ was removed together with a portion of the spermatic cord.

Fig. 7

Intraoperative image of the upper pole of the testis with an enhanced vascular pattern and bluish color. Two lesions resembling testicular appendages were found in the place where infiltration of the tunica albuginea had been suspected.

JoU-2016-0009-g007.jpg

The post-operative period passed without complications. The patient was discharged in a good general condition on the third day after the procedure. No abnormalities in wound healing were observed during a follow-up visit.

The following was found in a histopathological examination: “Macroscopically: a testis of 5 × 2 × 2 cm with an epididymis of 3 × 1 × 1 cm together with a portion of the spermatic cord 5 cm in length. Two nodules 0.3 cm in diameter are present on the outer surface. A cross-section of the testis reveals a grayish tumor of 1.2 × 1 × 1.5 cm. The tumor extends as far as the outer surface of the testis”.

Full pathomorphological diagnosis: Haemangioma capillare multifocale testis. Fibrosis testis et atrophia epithelii spermatogenici tubulorum seminiferorum maioris gradus. Immunohistochemistry: CK MNF116 (−), FVIII (−), CD31 (+), CD34 (+), vimentin (+), mesothelial cells (−), calretinin (−), MIB-1 = 8.4% (Fig. 8).

Fig. 8

Capillary hemangioma of the testis: A. benign tumor composed of a proliferation of capillary-size vessels in the testis parenchyma (hematoxylin and eosin, original magnification × 10); B. numerous capillary-size vessels lined by endothelial cells without anaplastic features (hematoxylin and eosin, original magnification × 40); C. Endothelial cells showing prominent immunostaining for CD31 (original magnification × 10); D. Endothelial cells showing prominent immunostaining for CD34 (original magnification × 20)

JoU-2016-0009-g008.jpg

Discussion

Hemangiomas are exceptionally rarely located in the testes. To date only 55 similar cases of this tumor have been reported, with only one series of cases involving 8 tumors(14).

Four histopathological types of testicular hemangiomas have been distinguished: cavernous, histiocytoid, capillary and papillary endothelial hyperplasia(2).

Capillary hemangioma of the testis is an exceptionally rare neoplasm in adults(2).

Due to the difficulties in differentiating between hemangiomas and malignant neoplasms of the testes on the basis of preoperative imaging scans the majority of patients undergo testis resection through the inguinal canal. The levels of routinely assayed tumor markers – AFP, beta-hCG and LDH are elevated only in one in two patients with a malignant neoplasm of the testis, while placental alkaline phosphatase (an optional marker) assay, which is used to monitor patients with a pure seminoma, is not recommended in smokers(5). If a malignant neoplasm of the testis is suspected, the standard course of action involves surgical exploration of the inguinal canal, isolation of the testis together with the tunica from the scrotum and cutting off the spermatic cord at the level of the internal inguinal ring. If there are doubts as to the malignancy of the tumor, an intraoperative histopathological examination of a sample collected from the lesion, or, even better, of the whole lesion removed together with a margin of healthy testicular parenchyma may be performed(4, 5). The guidelines of the European Association of Urology do not recommend testis-sparing resection if the other testis is normal, except for special circumstances. Partial resection of the testis whilst ensuring on-cological clearance may be performed if the volume of the lesion is smaller than 30% of the volume of the testis, metachronous or synchronous lesions occur in the contralateral testis, the testis operated on is the only one the patient has and the preoperative level of testosterone is normal. However, one needs to be prepared for the coexistence of testicular intraepithelial neoplasia (TIN) in as many 82% patients(5).

In an ultrasound scan malignant neoplasms of the testes usually take the form of focal, hypoechogenic lesions, although they may be characterized by a significant diversity. In the case of malignant lesions a significant part of the non-infiltrated testicular parenchyma has a preserved echogenicity and structure. In inflammation or post-traumatic conditions normal structure and echogenicity of the testis may sometimes be barely observed. Even if there is limited inflammation or focal hematoma, in the acute phase the remaining part of the testis usually has signs of edema. Malignant lesions may obscure the contours of the testes, while benign lesions may cause the testes to be enlarged, although without irregularities in the outer contours. The accompanying inflammation reaction and signs of epididymis edema usually indicate an inflammatory origin of testicular lesions(6, 7).

Reports on the use of magnetic resonance imaging in the diagnosis of capillary hemangiomas of the testes are rare. In the available descriptions T1- and T2-weighted imaging and the use of a contrast agent did not allow to determine whether the lesions were benign or malignant(8, 9).

In the present case physical examination and ultrasound assessment of the scrotum indicated a malignant neoplasm. Since the other testicle of the patient was not abnormal, a decision was made to remove the left testicle via the inguinal approach. One should bear in mind, however, that in selected patients with a small tumor of the only testis partial resection of the organ should be considered, especially if a Doppler ultrasound scan suggests hemangioma proliferation. In doubtful cases an intraoperative histopathological examination is recommended(10).

Conflict of interest

Authors do not report any financial or personal connections with other persons or organizations, which might negatively affect the contents of this publication and/or claim authorship rights to this publication.

References


  1. Kryvenko ON,Epstein JI,Testicular hemangioma: a series of 8 cases Am J Surg Pathol 2013 37 860 866
    [PUBMED] [CROSSREF]
  2. Mazal PR,Kratzik C,Kain R,Susani M,Capillary hemangioma of the testis J Clin Pathol 2000 53 641 642
    [PUBMED] [CROSSREF]
  3. Wong NC,Dason S,Pozdnyakov S,Alexopoulou I,Greenspan M,Capillary hemangioma of the testis: a rare benign tumour Can Urol Assoc J 2015 9 133 135
    [PUBMED] [CROSSREF]
  4. Mungan S,Turgutalp H,Ersöz S,Keskin F,Kutlu O,A rare neoplasm of the testis: capillary hemangioma Turk Patoloji Derg 2011 27 80 83
    [PUBMED]
  5. Albers P,Albrecht W,Algaba F,Bokemeyer C,Cohn-Cedermark G,Fizazi K,Guidelines on Testicular Cancer: 2015 Update Eur Urol 2015 68 1054 1068
    [PUBMED] [CROSSREF]
  6. Cochlin DL,Cochlin DL,Dubbins PA,Goldberg BB,Halpern EJ,The scrotum Urogenital Ultrasound 2006 London–New York Taylor & Francis 183 255
    [CROSSREF]
  7. Jakubowski W,Szopiński T,Sudoł-Szopińska I,Szopiński T,Moszna Diagnostyka ultrasonograficzna w urologii 2007 Warszawa–Zamość Roztoczańska Szkoła Ultrasonografii 129 153
  8. Essig M,Knopp MV,Hawighorst H,van Kaick G,MRI of capillary hemangioma of the testis J Comput Assist Tomogr 1997 21 402 404
    [PUBMED] [CROSSREF]
  9. Zaidi SN,Fathaddin AA,Testicular capillary hemangioma–a case report of a rare tumor Indian J Pathol Microbiol 2012 55 557 579
    [PUBMED] [CROSSREF]
  10. Passman C,Urban D,Klemm K,Lockhart M,Kenney P,Kolettis P,Testicular lesions other than germ cell tumours: feasibility of testis-sparing surgery BJU Int 2009 103 488 491
    [PUBMED] [CROSSREF]
XML PDF Share

FIGURES & TABLES

Fig. 1

Ultrasound scan. 12 MHz linear probe. Left testis. A lesion with a slightly decreased echogenicity is visible in its upper pole

Full Size   |   Slide (.pptx)

Fig. 2

Ultrasound scan. 12 MHz linear probe. Irregular contours of the upper pole of the left testis which suggest infiltration of the tunica albuginea

Full Size   |   Slide (.pptx)

Fig. 3

Ultrasound scan with color Doppler imaging. The vasculature of the upper pole of the left testis is distinctly larger than that of the remaining testicular tissue

Full Size   |   Slide (.pptx)

Fig. 4

Ultrasound scan with power Doppler imaging. The examination confirms a significantly larger blood flow in the upper pole of the testis in the area of the previously found lesion

Full Size   |   Slide (.pptx)

Fig. 5

3D ultrasound scan. Three-dimensional reconstruction of the left testis using color Doppler imaging

Full Size   |   Slide (.pptx)

Fig. 6

3D ultrasound scan. Three-dimensional reconstruction of the vasculature of the suspicious lesion located in the upper pole of the left testis allows for the determination of its precise topography

Full Size   |   Slide (.pptx)

Fig. 7

Intraoperative image of the upper pole of the testis with an enhanced vascular pattern and bluish color. Two lesions resembling testicular appendages were found in the place where infiltration of the tunica albuginea had been suspected.

Full Size   |   Slide (.pptx)

Fig. 8

Capillary hemangioma of the testis: A. benign tumor composed of a proliferation of capillary-size vessels in the testis parenchyma (hematoxylin and eosin, original magnification × 10); B. numerous capillary-size vessels lined by endothelial cells without anaplastic features (hematoxylin and eosin, original magnification × 40); C. Endothelial cells showing prominent immunostaining for CD31 (original magnification × 10); D. Endothelial cells showing prominent immunostaining for CD34 (original magnification × 20)

Full Size   |   Slide (.pptx)

REFERENCES

  1. Kryvenko ON,Epstein JI,Testicular hemangioma: a series of 8 cases Am J Surg Pathol 2013 37 860 866
    [PUBMED] [CROSSREF]
  2. Mazal PR,Kratzik C,Kain R,Susani M,Capillary hemangioma of the testis J Clin Pathol 2000 53 641 642
    [PUBMED] [CROSSREF]
  3. Wong NC,Dason S,Pozdnyakov S,Alexopoulou I,Greenspan M,Capillary hemangioma of the testis: a rare benign tumour Can Urol Assoc J 2015 9 133 135
    [PUBMED] [CROSSREF]
  4. Mungan S,Turgutalp H,Ersöz S,Keskin F,Kutlu O,A rare neoplasm of the testis: capillary hemangioma Turk Patoloji Derg 2011 27 80 83
    [PUBMED]
  5. Albers P,Albrecht W,Algaba F,Bokemeyer C,Cohn-Cedermark G,Fizazi K,Guidelines on Testicular Cancer: 2015 Update Eur Urol 2015 68 1054 1068
    [PUBMED] [CROSSREF]
  6. Cochlin DL,Cochlin DL,Dubbins PA,Goldberg BB,Halpern EJ,The scrotum Urogenital Ultrasound 2006 London–New York Taylor & Francis 183 255
    [CROSSREF]
  7. Jakubowski W,Szopiński T,Sudoł-Szopińska I,Szopiński T,Moszna Diagnostyka ultrasonograficzna w urologii 2007 Warszawa–Zamość Roztoczańska Szkoła Ultrasonografii 129 153
  8. Essig M,Knopp MV,Hawighorst H,van Kaick G,MRI of capillary hemangioma of the testis J Comput Assist Tomogr 1997 21 402 404
    [PUBMED] [CROSSREF]
  9. Zaidi SN,Fathaddin AA,Testicular capillary hemangioma–a case report of a rare tumor Indian J Pathol Microbiol 2012 55 557 579
    [PUBMED] [CROSSREF]
  10. Passman C,Urban D,Klemm K,Lockhart M,Kenney P,Kolettis P,Testicular lesions other than germ cell tumours: feasibility of testis-sparing surgery BJU Int 2009 103 488 491
    [PUBMED] [CROSSREF]

EXTRA FILES

COMMENTS