Scrotal massive lymphedema, also known as scrotal elephantiasis, represents a rare condition that accounts for ~0.6% of all cases of lymphedema (1-4). Although relatively common in areas with endemic filariasis, it is rarely observed in industrialized areas, where such a condition is almost exclusively iatrogenic or congenital (1-4). Scrotal massive lymphedema, is a disease usually caused by obstruction, aplasia, or hypoplasia of the lymphatic vessels draining the scrotum. It can be either congenital or acquired. The most common acquired etiology is infection due to lymphogranuloma venereum or filarial infestation with Wuchereria bancrofti. The scrotal skin usually becomes thickened and in severe cases, it can be affected by ulcerations (5). Scrotal elephantiasis can cause severe functional, social and emotional limitations due to recurrent skin infections, sexual dysfunction, pain and cosmetic deformity, as well as limitations of mobility and ambulation (1-4). The response to medical therapy is poor and the complete excision of the affected tissues, together with reconstructive methods can provide a definitive solution, irrespective of the etiology (1-4). The purpose of surgery is to excise the mass, to reconstruct the scrotum and repair the skin of the penis (6). Although a variety of surgical procedures have been reported, the ideal solution has not yet been identified and no definitive recommendations exist to date, at least to the best of our knowledge (1-4).

The present study describes the case of a Caucasian patient with massive idiopathic penoscrotal lymphedema. A novel surgical technique for scrotal elephantiasis correction was used for the treatment of this patient.

In May, 2020, a 43-year-old Caucasian male patient was admitted to the Department of Urology and Andrology of University Federico II of Naples (Naples, Italy) with massive scrotal elephantiasis. The past medical history of the patient was relevant for arterial hypertension. His previous surgical history included a circumcision at the age of 16. There was no history of sexually transmitted diseases, irradiation, or travel to endemic regions. The onset was spontaneous, and the swelling had substantially increased over the past 4 years. He reported psychological discomfort mainly due to the esthetic appearance of the genitalia and a burning sensation during micturition. No other notable findings were evident at the physical examination. The weight and height of the patient at the time of surgery were 140 kg and 190 cm, respectively (body mass index, 38.7 kg/m²; obesity class II). He presented a massive swelling of the scrotum with cutaneous indurations covering the vast majority of the area (Fig. 1). His penis was completely buried, and the glans was not visible. The penis, the testes and the spermatic cords were not palpable. Laboratory testing (including human immunodeficiency virus, markers for testicular cancer, antibodies to schistosomes, Chlamydia trachomatis and filariae) were negative. Magnetic resonance imaging of the abdomen revealed the integrity of the corpora cavernosa, the spermatic cord, as well as the testis. On the other hand, the status of the glans was uncertain. The patient was thus scheduled for a complete scrotectomy. The various stages of surgery included the following:

The management of penoscrotal elephantiasis should be focused on treating the underlying causes. A neoplastic process has to be excluded and medical treatment should be administered when necessary. To complete the treatment a surgical approach is essential. There are several ways to approach such conditions (7-9). In such cases, an excisional approach is the only possible treatment. Several surgical techniques have been presented over the years. In 2008, Garaffa et al (10) described the ‘Scrotal inverted ‘W’ incision’. In 2014, Machol et al (11) demonstrated that lateral-based scrotal flaps (with or without mid-raphe Z-plasty) allowed for a correct anatomical reconstruction. In addition, in 2018, Irdam and Fadhly (12) described a circular incision encircling the scrotum.

Furthermroe, in 2019, Alnajjar et al (13) suggested an innovative approach with a ‘batman incision’ that avoids the need for pedicled flaps and their associated risks of flap ischemia/necrosis, longer surgical times and additional donor site wounds. On the other hand, such technique requires penile transposition and lower abdomen extensive mobilization (13).

The present study ‘redrew’ the surgical approach to this uncommon disease using a ‘hanger-shaped incision’ in order to obtain a trapezoidal cavity. Such an incision permits the formation of neo-raphe, neo-septum and eventually, the neo-scrotum as a method with which to restore as much as possible the natural appearance of the genitalia. The neo-septum was designed in order to avoid the risk of spermatic cords twisting. In fact, the continuous stretching of the tissues, due to the weight of the mass, could irremediably cause a lengthening of the cords. When approaching such large masses, a top-down approach is suggested, as the identification of the noble structures becomes more feasible and safe. Moreover, the ‘hanger-shaped incision’ permits a T-closure of the wound, preventing the extensive mobilization of the pre-pubic area which will be mandatory for a penile transposition. In addition, a penile transposition requires a perfect ring excision to avoid either difficulty with the grafting or constriction during erection. On the other hand, the T closure ensures a redistribution of the tensile strengths on three different unconnected flaps (Figs. 11 and 12).

In conclusion, in the case described in the present study, the ‘hanger-shaped incision’ minimized the use of rotation flaps and should reduced the risk of recurrence by eliminating all the cellulitis tissue, restoring the natural compartmentalization of the scrotum and improving the esthetic appearance.