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Revista argentina de cirugía

Print version ISSN 2250-639XOn-line version ISSN 2250-639X

Rev. argent. cir. vol.115 no.3 Cap. Fed. Aug. 2023

http://dx.doi.org/10.25132/raac.v115.n3.1649 

Articles

Giant scrotal lymphedema

Jorge A. Obeid1 

Rodolfo Benech1 

Gustavo E. Gauna1 

Mauricio A. Minetti1 

Federico N. Gudelj1 

Agostina Cogliano1  * 

1 Servicio de Cirugía General del Hospital J. B. Iturraspe, Ciudad de Santa Fe, Argentina.

Scrotal lymphedema (SL) is the accumulation of lymphatic fluid in the interstitial space of the skin and subcutaneous tissue due to reduced lymphatic drainage, resulting in enlargement of the penis and scrotum. Chronic cases may also involve fibrosis and deposition of fat1. The etiologies are multiple and can be classified into two groups: a) congenital diseases, which represent approximately 15%, and, b) acquired conditions, including neoplasms, infections, obesity, granulomatous diseases, fluid balance disorders, and idiopathic diseases2. The main cause of SL worldwide is lymphatic involvement due to filariasis, an infection caused by nematode parasites classified as nematodes of the family Filariodidea, and transmitted by vectors, either mosquitoes or other arthropods3.

This physical condition represents an uncomfortable clinical situation for patients, particularly when it results in large deformities known as elephantiasis with major functional, psychological and social repercussions for the patients. We report the case of a 43-year-old male patient, with morbid obesity [body mass index (BMI) 52.57 (143 kg/165 cm)], and no medical or surgical history, who was referred from a primary health care center for presenting a large inguino-scrotal mass for 20 years that interfered with ambulation and evacuation habits.

The patient was confined to bed with severe depression, a situation that he described as desperate. He had previously consulted in other institutions without receiving any response to his problems. On physical examination, he presented a bulky inguino-scrotal mass that reached the floor in the standing position. The external genitalia were buried within the mass, and there was a midline orifice on the anterior surface could correspond to a fistulous tract communicating with the penis. The skin presented hyperkeratosis, ochre dermatitis and hyperpigmentation. There were also multiple varicose veins in the lower limbs. The patient complaint of serious mobility impairment that interfered with his hygiene habits.

The complementary tests showed slight renal function impairment (creatinine level 1.66 mg/dL) and the computed tomography (CT) scan demonstrated an enlarged scrotal sac with thickened soft tissues (probably due to edema), measuring approximately 68 cm in length × 51 cm (transverse diameter) × 32 cm (anteroposterior diameter). The sac content could not be identified. The case was discussed in the surgical case conference with the participation of general surgeons, urologists, and plastic and reconstructive surgeons. The approach decided was resection and reconstructive surgery. Under general and spinal anesthesia, the scrotal edematous mass mainly made up of fibrotic tissue was resected; the external genitalia were carefully dissected, adequate hemostasis was checked and the urinary tract was protected with a urinary catheter. Finally, the penis was reconstructed and the perineal floor was repaired with advancement flaps. The testes were left in the subcutaneous tissue. A latex drain was placed in the subcutaneous tissue (Fig. 1). The pathology report demonstrated histological findings consistent with massive localized scrotal lymphedema. We could verify that the therapeutic patient-focused and centered targets were met, as the giant scrotal edema (62 kg) was completely resected.

Figure 1 The black arrows indicate the testes. 

After surgery, the patient was immediately admitted to the intensive care unit where he remained for 7 days and when then transferred to the general ward. He was discharged on postoperative day 21 due to favorable progress with indications of strict controls and kinesiotherapy. He was also followed up by mental health physicians, nutritionists and the Obesity Committee. She is currently on schedule for dermolipectomy (Fig. 2). Scrotal lymphedema is a rare entity due the accumulation of lymphatic fluid in the subcutaneous tissue. It is classified as primary, caused by developmental abnormalities of the lymphatic system, or secondary to disruption or obstruction of the lymphatic vessels produced by trauma, radiotherapy, neoplasm, surgery, infection or idiopathic causes4. In our society, morbid obesity (BMI > 40 kg/m2) is a type of idiopathic secondary lymphedema, as the body habitus with large and heavy folds can directly compress the underlying lymphatic vessels5. The progressive swelling of lymphedema is often soft with pitting edema at onset followed by chronic inflammation and fibrosis, presenting as nonpitting edema. In later stages, the skin appears with hyperkeratosis and vesicles due to lymph fluid overflow. Bacterial infection through the breakdown of the physical defenses of the skin and a proteinrich growth media for bacterial proliferation lead to frequent episodes of cellulitis6.

Figure 2 Preoperative and postoperative images. 

This entity affects both the emotional and physical spheres. In addition to the grotesque aspect, the progression of the condition impairs ambulation, makes sexual intercourse impossible, and impairs both voiding in the standing position and proper hygiene of the perineal region with subsequent body odor and recurrent episodes of skin infections causing severe damage to the patient’s quality of life5,6.

Treatment depends on the stage and severity of patient’s presentation. In mild acquired cases treatment involves management of the cause, while in congenital cases conservative treatment is indicated. Surgery is the treatment of choice for severe cases. Surgical management requires resection of the lymphedematous tissue and reconstruction of the scrotum with grafts, fasciocutaneous or musculocutaneous thigh flaps to cover the testes or expansion of adjacent tissues3,4,6.

In conclusion, giant SL is a very rare entity, which causes major impairment in patients’ quality of life, affects biopsychosocial well-being and requires a multidisciplinary approach by a team of qualified professionals comprising clinicians, general surgeons, plastic surgeons, urologists, psychologists, nutritionists, kinesiologists and social workers. Effective treatment, either medical or surgical in severe cases, can achieve satisfactory results, allowing patients to improve hygiene habits, self-esteem, sex life, posture and ambulation, and thus provide them with tools for returning to social life and work.

Referencias bibliográficas /References

1. Miranda H, Colangelo AC, Antunes M, Schiavone M, Merigliano S, Pizzol D. Giant elephantiasis and inguino-scrotal hernia. PLoS Negl Trop Dis 2017;11(6): e0005494. doi.org/10.1371/journal.pntd.0005494 [ Links ]

2. Sánchez JD. PAHO/WHO [Internet]. Paho.org. [cited 2021 May 4]. Disponible en: Disponible en: https://www.paho.org/hq/index.php?option=com_content&view=arti%20cle&id=5855:2011-%20general-information-lymphatictLinks ]

3. Hornberger BJ, Elmore JM, Roehrborn CG. Idiopathic scrotal elephantiasis. Urology 2005;65(2):389. doi: 10.1016/j.urology.2004.08.040. [ Links ]

4. Sánchez-Alvarado JP, Procuna-Hernández JN, Manzanilla-García HA, Gutiérrez-Godínez FA, Rosas-Ramírez A. Linfedema primario escrotal gigante (enfermedad de Meige): reporte de 2 casos [Internet]. Medigraphic.com. 2008 [cited 2021 May 4]. Disponible en: Disponible en: https://www.medigraphic.com/pdfs/uro/ur-2008/ur086i.pdfLinks ]

5. Vargas-Mamani JH, Torrez-Miranda SC, Campos Mendoza RE, Torrico Castillo JN, Dávalos Grágeda MK. Intervención quirúrgica de linfedema escrotal gigante, Bolivia. Rev Cienc Cienc Méd 2018; 21(2): 60-66. Disponible en: <http://www.scielo.org.bo/scielo.php?script=sci_arttext&pid=S1817-74332018000200011&lng=es &nrm=iso>. ISSN 2077-3323. [ Links ]

6. Thejeswi P, Prabhu S, Augustine AJ, Ram S. Giant scrotal lymphoedema - A case report. Int J Surg Case Rep 2012; 3(7): 269-271. [ Links ]

Received: November 19, 2021; Accepted: February 16, 2022

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