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  • Remedy Publications LLC., | http://anncaserep.com/

    Annals of Clinical Case Reports

    2016 | Volume 1 | Article 11771

    Introduction A hydrocele in the inguinoscrotal region is most frequently the result of an inguinal and/

    or scrotal cyst. A differential diagnosis of lymphangioma in the inguinal and/or scrotal region is difficult because of its rarity [1] however, a physician should be aware of such a diagnosis when initiating treatment of a hydrocele. Any lymphangioma found should be completely excised to prevent a recurrence [2]. However, an accurate diagnosis is essential for such radical treatment. We report here an illustrative case of a scrotal lymphangioma with the pathognomonic symptom of intracystic bleeding and subsequent, essential diagnostic imaging.

    Case Presentation A healthy boy, aged three years and eight months, was referred to our institute because a

    swelling in his right scrotum had enlarged; the scrotal skin had also changed to a blue color without any apparent reason. His right scrotum had been swelling since he was six months old, and this was consequently diagnosed as a hydrocele.

    An ultrasound examination of the swollen scrotum revealed a cystic lesion, 20 × 30 mm in size, which extended from the inguinal area to the scrotum. We diagnosed a hydrocele testis by gray scale ultrasonography without color Doppler. An inguinal approach for an operation was selected, which subsequently revealed a multilobulated cyst in a subcutaneous region of the inguinal area that extended to the scrotum. The cyst had a slightly brownish color. From this finding, we concluded that this cyst was not a hydrocele but a congenital cystic lesion. The cyst was subsequently completely resected. After the operation, we reviewed the ultrasonography and identified a trabecular component inside the cyst (Figure 1A and B). A subsequent histological examination revealed that this cyst was composed of a thickened fibrous wall and an inner epithelial layer, which was positive for the lymphoepithelial antigen, D2-40 (Figure 2A and B). As a result, the final pathological diagnosis was a cystic-type lymphangioma, with a relatively large cyst bordering the septum.

    Discussion Lymphangioma has been classified into three subtypes, such as capillary, cavernous

    lymphangiomas and cystic hygroma. This classification is based on their microscopic characteristics. Lymphangiomas occur with about a 6% frequency in benign tumors [3]. Superficial lymphangiomas

    Scrotal Lymphangioma: Symptoms and Diagnosis

    OPEN ACCESS

    *Correspondence: Makoto Komura, Department of

    Pediatric Surgery, Saitama Medical University, 7-3-1 Hongo, Bunkyo-ku,

    TOKYO, 113-8655 Japan, Tel: +81-3- 3815-5411; Fax: +81-3-5800-5104;

    E-mail: [email protected] Received Date: 16 Sep 2016 Accepted Date: 14 Oct 2016

    Published Date: 03 Nov 2016

    Citation: Komura M, Tobari S, Kanamori Y, Kato

    M, mi KK, Hoshiai R, et al. Scrotal Lymphangioma: Symptoms and

    Diagnosis. Ann Clin Case Rep. 2016; 1: 1177.

    Copyright © 2016 Komura M. This is an open access article distributed under

    the Creative Commons Attribution License, which permits unrestricted

    use, distribution, and reproduction in any medium, provided the original work

    is properly cited.

    Case Report Published: 03 Nov, 2016

    Abstract We report here in a case of a scrotal lymphangioma with the characteristic symptom of intracystic bleeding that occurred in the absence of any traumatic episode. A boy, aged three years and eight months, presented with a swelling in his inguinoscrotal region that had gradually grown over time. By three years of age, the skin color of the scrotum had taken on a bluish tinge. Exploratory surgery revealed a multi lobular cyst in the subcutaneous region of the inguinal area that had spread to the scrotum. The cystic fluid was serous but slightly brownish. The inner epithelial layer of the cyst was positive for the lympho epithelial antigen, D2-40, indicating a lymphangioma. In conclusion, a cyst in the inguinoscrotal region that shows hemorrhaging in the absence of pain indicates this to be a lymphangioma.

    Keywords: Lymphangioma of scrotum; Hemorrhage; Hydrocele; Color doppler sonography

    Makoto Komura1,5*, Shoichi Tobari2, Yutaka Kanamori5, Masahisa Kato2, Kazuo Koizu mi3, Ryuji Hoshiai1, Jyunji Shiga4 and Katsunori Nishida2

    1Department of Pediatric Surgery, Tsudanuma Chuo General Hospital, Japan

    2Department of Surgery, Tsudanuma Chuo General Hospital, Japan

    3Department of Urology, Tsudanuma Chuo General Hospital, Japan

    4Department of Pathology, Tsudanuma Chuo General Hospital, Japan

    5Department of Pediatrics, The University of Tokyo Hospital, Japan

  • Makoto Komura, et al. Annals of Clinical Case Reports - Urology: Case Reports

    Remedy Publications LLC., | http://anncaserep.com/ 2016 | Volume 1 | Article 11772

    are often noticed in children, with the most commonly affected sites being the neck (75%) and maxilla (20%) [4]. Of these, inguinal or scrotal lymphangiomas have rarely been reported, with only about 80 cases described in the English medical literature to date [2,5]. In contrast, hydroceles occur quite frequently, often as the result of an inguinal and/or scrotal cyst [5]. However, distinguishing between a hydrocele and a scrotal lymphangioma based only on physical findings is quite difficult. In particular, a scrotal lymphangioma is often misdiagnosed as a hydrocele, inguinal hernia, hematocele, varicocele or possible torsion of the testis because of its rarity [1].

    In general, a lymphangioma located anywhere in the body can grow to a large size as a result of infection or hemorrhage [6]. The incidence of hemorrhage in scrotal and/or inguinal lymphangiomas is approximately 30%, calculated from 33 reported cases of scrotal or inguinal lymphangiomas in English language medical papers published since 1964 [2,7-10]. A cyst with an obvious hemorrhage that is located in the inguinal and/or scrotal region is a characteristic sign of a scrotal and/or inguinal lymphangioma, with the hemorrhage allowing us to make a differential diagnosis of a lymphangioma.

    We should, therefore, consider a lymphangioma when making a differential, routine diagnosis of a hydrocele in the inguinal and/ or scrotal region. Ultrasonography with color Doppler as a routine examination should be performed to image the blood supply in the lymphangioma wall [8]. In addition, magnetic resonance imaging can

    A

    B

    Figure 1: (A) Longitudinal image: A low echoic area around the right testis was visualized by ultrasonography. A septal wall was revealed in this routine ultrasonography examination. (B) Cross-sectional image: An ultrasound figure indicates a normal testis, with the fluid-filled lesion appearing similar to a hydrocele.

    A

    B

    Figure 2: (A) The cyst displayed lobules of various sizes in a histological section. Microscopically, the wall of the cyst consisted of fibrotic tissue (H&E staining). (B) The inner epithelial surface of the cyst was stained by anti–D2- 40 antibody, a lymphoepithelial-specific marker (brown stain) in a histological section. Cell nuclei were counterstained with hematoxylin.

    clearly show the exact region of the cyst, with characteristic findings of the wall [2].

    In conclusion, the appearance of a hemorrhage in a scrotal and/or inguinal cyst in the absence of pain should identify this as a lymphangioma.

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    2. Hamada Y, Yagi K, Tanano A, Y. Kato K. Takada K. Sato M, et al. Cystic lymphangioma of the scrotum. Pediatr Surg Int. 1998; 13: 442-444.

    3. Enjolras O, Wassef M, Chapot R. Color atlas of vascular tumors and vascular malformations. New York: Cambridge University Press; 2007.

    4. Singh S, Baboo ML, Pathak LC. Cystic lymphangioma in children: Report of 32 cases including lesions at rare’ sites. Surgery. 1971; 69: 947-951.

    5. Gueukdjian SA. Lymphangioma of the groin and scrotum. J Inter College Surg. 1955; 24: 159-170.

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  • Makoto Komura, et al. Annals of Clinical Case Reports - Urology: Case Reports

    Remedy Publications LLC., | http://anncaserep.com/ 2016 | Volume 1 | Article 11773

    al. Role of Doppler sonography in the diagnosis of cystic lymphangioma of the scrotum. J Clin Ultrasound. 2002; 30: 384-387.

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