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LETTER TO EDITOR  
Year : 2023  |  Volume : 66  |  Issue : 1  |  Page : 222-225
Testicular hemangioma in a child: A rare case report and review of literature


Department of Urology, Anhui Provincial Children's Hospital and Children's Hospital of Anhui Medical University, Hefei, Anhui, P. R. China

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Date of Submission01-Sep-2021
Date of Decision17-Oct-2021
Date of Acceptance02-Dec-2021
Date of Web Publication18-Jan-2023
 

How to cite this article:
Zhang K, Zhang Y, Chao M. Testicular hemangioma in a child: A rare case report and review of literature. Indian J Pathol Microbiol 2023;66:222-5

How to cite this URL:
Zhang K, Zhang Y, Chao M. Testicular hemangioma in a child: A rare case report and review of literature. Indian J Pathol Microbiol [serial online] 2023 [cited 2023 Feb 7];66:222-5. Available from: https://www.ijpmonline.org/text.asp?2023/66/1/222/367999




Dear Editor,

Testicular hemangioma are rare benign vascular tumors that arise from the inner layer of the tunica albuginea.[1] It was commonly seen in infants and children without any discomfort. Some cases are poorly defined and indistinguishable from malignant tumors. To date, only a few studies were reported on testicular hemangioma in the literature. We herein present the case of a 3-year-old male patient with hydrocele accompanying right testicular hemangioma. Then, testicular-sparing surgery can be performed for definitive treatment in patient. He is recovered well after 3 months of follow-up. This article discusses the diagnosis and currently available treatment options of testicular hemangioma.

We reported the case of a 3-year-old male patient with hydrocele accompanying right testicular hemangioma. Physical examination revealed a painless scrotal mass with a positive transillumination test. Initial laboratory studies including urinalysis, urine culture, complete blood count, and so on were negative. The levels of preoperative serum tumor markers are normal. An ultrasound scan of the scrotum revealed a solid mass located in the upper pole of the right testis, 19 mm in diameter with slightly decreased echogenicity and regular contours [Figure 1]a. Doppler scans demonstrated a dense network of blood vessels and increased blood flow in the lesion described [Figure 1]b. The patient underwent laparscopic exploration, and then open surgery was performed through an inguinal approach. Taking the suspected malignancy into account, a decision was made to remove the solid mass using the inguinal approach. The specimen was sent to do a high-speed frozen section histodiagnosis and found it was hemangioma [Figure 2]. In order to preservation of a healthy tissue margin, resection of the solid mass was performed in patient. Then, the wound was stitched up. Histopathological examination of the whole specimen revealed a multifocal hemangioma of the testis accompanying proliferation. Immunohistochemistry: vimentin (+), CD31 (+), CD34 (+), Ki-67(+8%), CK(-), and calretinin (-) [Figure 3]. The patient was discharged on day 4 after surgery, and recovered well after postoperative 3 months. Bilateral scrotal hydrocele was found at the age of 2 in the patient's medical history, and the ligation of peritoneovaginal duct was performed by laparoscopic technique.
Figure 1: Testicular hemangioma in a 3-year-old male child. (a) Ultrasound images of the right testicle show a hypoechoic intratesticular mass, measuring 1.9 × 1.4 × 1.2 cm. (b) Color Doppler ultrasound image of the right testicle demonstrates blood flow within the intratesticular mass

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Figure 2: Hematoxylin and eosin staining revealed a proliferation of capillary-size vessels in the testis parenchyma (original magnification × 100)

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Figure 3: Immunohistochemistry of capillary hemangioma of the testis. (a) Endothelial cells showing prominent immunostaining for vimentin (original magnification × 200); (b) endothelial cells showing prominent immunostaining for CD31 (original magnification × 200); (c) endothelial cells showing prominent immunostaining for Ki-67 (original magnification × 200); and (d) endothelial cells showing prominent immunostaining for CD34 (original magnification × 200)

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Testicular tumors are predominantly malignant in adults. In some cases, it is difficult to discriminate benign tumor from malignancy in terms of the diagnosis and treatment. Generally, radical orchidectomy remains the best choice for patients with malignancy, whereas treatment option of benign testicular tumors is the testis-sparing approach. The preoperative differentiation is important for clinicians who encounter a testicular mass. Testicular hemangiomas in children are extremely rare. As shown in [Table 1], we found that testicular hemangiomas occur in diverse age groups.[2],[3],[4],[5],[6],[7] Hemangioma is a rare benign tumor of the testicle that arise from the inner layer of the tunica albuginea, which often be confronted with a significant diagnostic and therapeutic dilemma.[1] This may be in part because testicular hemangioma have not been extensively reported in the literature. In addition, pediatric testicular tumors are distinct from adult testicular tumors. Whereas only a small number of adult testicular tumors are benign, approximately one-third of pediatric testicular tumors are benign.[8] That suggests that prepubertal tumors should be managed in a manner different from adult. We present this rare case in a child and discuss the diagnosis and currently treatment options of testicular hemangioma.
Table 1: Main characteristics of the recent studies

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Most testicular hemangiomas present as painless but palpable lesions similar to malignant tumors on clinical presentation. Appropriate preoperative and intraoperative recognition of tumors is vital for treatment options. From [Table 1], ultrasonography is a commonly useful tool to diagnose hemangioma in clinic. Testicular hemangioma often presents as hypoechogenic central part with hyperechogenic findings. Although ultrasonography has been applied successively to the diagnose of testicular hemangioma, surgical exploration and pathology are still required for definitive diagnosis. Occasionally magnetic resonance imaging (MRI) could distinguish benign tumors from malignant testicular tumors. However, reports on the use of MRI in the diagnosis of testicular hemangiomas are rare. T1- and T2-weighted images did not allow to determine whether the tumor was benign or malignant. Clinical examination and imaging studies do not often suffice to get a correct diagnosis. Histological examination with immunohistochemical staining contributes to making a correct and ultimate diagnosis. Serum tumor markers are of utmost value for diagnosis as well as for prognosis. The knowledge of preoperative serum tumor markers could avoid a discrepancy in the pathology report. Routinely assayed tumor markers, including lactate dehydrogenase, β-human chorionic gonadotropin (hCG), and α-fetoprotein (AFP) are usually normal in children with a hemangioma of the testis. Immunohistochemically, endothelial (CD31, CD34, factor VIIl-related antigen, vascular endothelial growth factor) or mesenchymal (vimentin) markers have previously been reported as positive, which is helpful for differential diagnosis.

As for testicular malignancy, the standard course of surgery mainly includes, via the inguinal approach, cutting off the spermatic cord at the level of the internal inguinal ring. Further management of the patient is dependent on the diagnosis at microscopy. If you make doubt for malignancy of the testis, intraoperative frozen sections may be recommended from the lesion, or the whole lesion together with a margin of healthy testis. If the diagnosis is not definitive during operation, orchidectomy must be performed. If a benign lesion is confirmed intraoperatively, testicular-sparing surgery can be performed for definitive treatment instead of a radical orchiectomy. Of course, we should take some important data such as patient demographics, age, laterality, the type of specimen, the anatomical origin of lymph nodes, and histopathology of any prior excised testicular tumors into consideration. It should be noted that anyone should carefully consider all the details before diagnose and treatment.[9]

Prepubertal testicular tumors are distinct from those of adults in histologic characteristics, molecular biology, and clinical behavior. Current therapeutic modalities of testicular hemangioma include surgical excision, laser fulguration, intralesional sclerotherapy, and cryotherapy. From [Table 1], complete local excision and follow-up are widely used for testicular hemangioma. Because intraoperative frozen section examination could be performed, testicular sparing surgery is possible, especially for patients with negative tumor marker findings. Meanwhile, in order to protect fertility, we also should take testicular sparing surgery into consideration first. Testicular sparing surgery appears to be a feasible option for benign tumors and is safe and efficacious in long-term follow-up.[10] Local recurrence occurs in some patients but there are no reports of distant metastases. To date, testicular hemangiomas, managed by either radical or partial orchiectomy, have not yet shown any malignant potential. Sometimes the rapid growth of the testicular hemangioma has been previously reported and might be explained by the development of wide, intratumoral hemorrhage.[10]

Testicular hemangiomas are rare benign vascular tumors. We performed testicular-sparing surgery in our case. In this way, we aimed to protect the baby from hypogonadism, infertility, and poor cosmesis. However, long-term follow-up is needed.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Ghoreifi A, Djaladat H. Management of primary testicular tumor. Urol Clin North Am 2019;46:333-9.  Back to cited text no. 1
    
2.
Kutsal C, Baloğlu İH, Albayrak AT. Hydrocele accompanying testicular cavernous hemangioma: A infant case report. Int J Surg Case Rep 2021;82:105844.  Back to cited text no. 2
    
3.
Li F, Han S, Liu L, Xu S, Cai D, Liang Z, et al. Benign testicular cavernous hemangioma presenting with acute onset: A case report. Mol Clin Oncol 2020;13:19-22.  Back to cited text no. 3
    
4.
Tepeneu NF, Krafka K, Meglic S, Rogatsch H, Fasching G. Testicular cavernous hemangioma associated with testicular torsion-Case report and review of literature. Int J Surg Case Rep 2018;49:247-50.  Back to cited text no. 4
    
5.
Yu D, Lei Y. Testicular hemangioma: A case report. Urol Case Rep 2017;16:20-1.  Back to cited text no. 5
    
6.
Białek W, Rudzki S, Wronecki L. Capillary hemangioma of the testis. A case report of a rare benign tumor. J Ultrason 2016;16:87-93.  Back to cited text no. 6
    
7.
Ben Abda R, Bess D, Nieves-Robbins N. Testicular hemangioma mimicking a malignant neoplasm. Radiol Case Rep 2016;11:121-3.  Back to cited text no. 7
    
8.
Lee SD. Korean Society of Pediatric Urology. Epidemiological and clinical behavior of prepubertal testicular tumors in Korea. J Urol 2004;172:674-8.  Back to cited text no. 8
    
9.
Ambekar A, Rao V, Pai SA, Bindhu MR, Midha D, Kaushal S, et al. Grossing and reporting of testicular tumor specimens: An evidence-based approach. Indian J Cancer 2020;57:7-12.  Back to cited text no. 9
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10.
Nerli RB, Ajay G, Shivangouda P, Pravin P, Reddy M, Pujar VC. Prepubertal testicular tumors: Our 10 years experience. Indian J Cancer 2010;47:292-5.  Back to cited text no. 10
[PUBMED]  [Full text]  

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Correspondence Address:
Min Chao
Department of Urology, Anhui Provincial Children's Hospital and Children's Hospital of Anhui Medical University, No. 39 Wangjiang East Road, Hefei 230051, Anhui Province
P. R. China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpm.ijpm_881_21

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