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

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

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

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

Fig. 5

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

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

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.

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)

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^(1–4).

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⁾.