Scrotal lymphangioma in a middle-aged man

Abstract

Scrotal lymphangioma is a rare, acquired, idiopathic conditions. Surgical resection can result in successful outcomes. A 43-year-old man presented with a painless scrotal mass. Ultrasonography and magnetic resonance imaging revealed a multifocal cystic mass in the scrotum that showed partial hypervascularity without connection to the skin. We performed complete surgical resection of the mass and histopathologically diagnosed the patient with lymphangioma. Acquired scrotal lymphangiomas are rare. Moreover, there was no apparent cause, such as injury; thus, this case was considered idiopathic. Complete surgical resection led to a successful resolution.

Abbreviations

MRI

Magnetic resonance imaging

US

Ultrasound

1. Introduction

Most lymphangioma are observed in neonates or infants as congenital malformations occurring in the neck and axilla. We report a rare case of idiopathic acquired lymphangioma in an adult patient. This is rare idiopathic acquired case in an adult patient. Acquired lymphangioma often has episodes, such as injury or infection; however, this case had no episodes due to lymphangioma.

2. Case presentation

A 43-year-old male patient was referred to our institution because of a painless mass in the scrotum. The scrotal mass was located in the midline of the scrotum and was approximately 3 cm in size, elastic, soft, and had poor mobility (Fig. 1). Ultrasonography revealed normal bilateral testes and a multifocal cystic mass immediately below the scrotal skin. The inferior of the mass was mosaic in shape. Hypervascularization was observed in some parts of the mass (Fig. 2). Magnetic resonance imaging (MRI) was also performed. It suggested that the mass was derived from the scrotal septum or testicular sheath membrane (Fig. 3). We did not observe a connection between the mass and the bilateral testes using either ultrasound or MRI.

Fig. 1.

Gross finding of the scrotal area.

A mass exists in the middle of the scrotum.

Fig. 2.

Ultrasonography of the scrotum.

A multifocal cystic mass is observed, with rich blood flow in some areas.

Fig. 3.

Gadolinium-enhanced magnetic resonance image of sagittal view.

A multifocal cystic mass with a clear boundary with the normal testis is observed.

Tumor markers of testicular carcinomas, such as LDH, α-fetoprotein, and hCG-β, were negative. The scrotal mass was surgically resected by incising the scrotal suture line. Although there was firm adhesion between the mass and the meatus, it was possible to separate the scrotal mass without injuring the bilateral testes and urethra. Histopathological findings revealed the presence of a dilated cystic structure inside the abundant smooth muscle that appeared to be a lymphatic vessel. Thus, the patient was diagnosed with lymphangioma of the scrotum. The surgical margins revealed a negative pathological lesion. The surgical resection was considered successful. The patient progressed postoperatively without any evidence of recurrence.

The patient provided informed consent for publication and the study was approved by the relevant ethical committee.

3. Discussion

Here, we describe a case of scrotal lymphangioma in an adult patient who underwent surgery. Most lymphangiomas are congenital malformations in the neonatal or infant period, and 95% occur in the neck and axilla. Other geneses, such as the mediastinum, mesentery, retroperitoneum, viscera, and bone, have been reported. Secondary lymphangiomas are reportedly caused by infection, trauma or postsurgical lymphatic blockage.¹ This patient did not have surgical or traumatic episodes or infectious history as risk factors. Thus, this was considered a rare adult idiopathic case.

There are two types of lymphangiomas: simple (capillary) and cavernous (cystic hygroma) type. The pathological findings of the lymphangioma were dilated lymph vessels with lined with flat endothelial cells, mainly in the upper dermis and subcutis.² In this case, the vascular structures disappeared, and dilated cystic structures derived from the lymphatic vessels were abundant within the smooth muscle. Thus, it was considered a pathologically cavernous-type lymphangioma.

The differential diagnosis included a multifocal cystic tumor with hemorrhage, or an intra-scrotal extratesticular mass derived from the scrotal septum or testicular sheath. Imaging diagnosis is useful in a non-invasive manner. Complex cystic lesions were observed by ultrasonography (US). MRI demonstrated multiloculated, hyperintense areas on T2-weighted images. Furthermore, T2-weighted images have a good sensitivity for evaluating the presence of tumors far from the epidermis.¹ Puncture of the cystic lesion can help in histopathological diagnosis; but it is not an adequate technique for treatment and had a risk of dissemination.³

Treatment procedures include embolization, laser therapy, electrocoagulation, cryotherapy, radiation, sclerotherapy, and surgical excision. No treatment procedure has high-level evidence; however, most studies report that the most definitive treatment is surgical resection. The most common postoperative complication is lymphangioma recurrence, with 25%–50% incidence due to incomplete resection.² Other reported complications of surgical resection include scrotal edema, lymphatic leakage, and local infection. Saade et al. reported a case of successful laser treatment. They suggested that fewer sessions and less exposure to the laser prevented complications, such as postoperative pain and cosmetic appearance with white scar-like lesions.⁴ In the other cases, transcatheter lymphatic sclerotherapy and embolization were performed. Kang et al. performed sclerotherapy using bleomycin A5 with embolization of ethiodized oil. The authors suggested that multiple rounds of sclerotherapy are required for large lymphangioma.⁵ We performed surgical resection to avoid dissemination if the tumor had malignant potential and achieved a complete cure. No recurrence was observed after resection.

4. Conclusion

Here, we reported a case of acquired idiopathic scrotal lymphangioma that was successfully surgically resected without recurrence. Lymphangiomas are rare in adult. Most adult cases have episodes, such as scrotal injury and infections; however, there were no such episodes in this case.

Funding

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Declaration of competing interest

None.