Case Report
Scrotal Lymphangioma: Symptoms and Diagnosis
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
*Corresponding author: Makoto Komura, Department of Pediatric Surgery, Saitama Medical University, 7-3-1 Hongo, Bunkyo-ku, TOKYO, 113-8655 Japan
Published: 03 Nov, 2016
Cite this article as: 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.
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
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.
Figure 1
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.
Figure 2
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.
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 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 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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