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We are presenting the case of a 20 year-old Haitian man, living in the south east part of Haiti, with gigantic scrotal and right leg elephantiasis. 

Keywords: giant scrotal elephantiasis, Wuchereria bancrofti
Corresponding Author: Jacques M. Jeudy, MD, Urology/Transplant Surgery. Haiti Transplant Program. Centre Hospitalier du Sacre Coeur, 34 Ave Charles Sumner, Port–au-Prince, Haiti, Ht 6114. Email: haititransplant@yahoo.com

Introduction

We are presenting the case of a 20 year-old Haitian man, living in the south east part of Haiti, with gigantic scrotal and right leg elephantiasis. He was diagnosed with a confirmed filariasis in 2003 at St Croix hospital of Leogane, 20 miles from Port-au-Prince. The city of Leogane is officially known as an endemic area for Wuchereria bancrofti infestation. Despite standard medical management, good skin care and compression bandaging, the disease has slowly but steadily progressed. Over a five year period, the patient has become almost completely incapacitated by a huge swelling of the external genital organs and his right leg. This particular case of scrotal elephantiasis ranks among the biggest ever recorded throughout our review of medical literature.  

Case Report

Otherwise, he has been healthy for the most part, except for complaint of a persistent anemia and, at times, some abdominal pains that were not further investigated. There was no history of high blood pressure or diabetes mellitus, and no traumatic injury. Renal and liver function was normal. At physical examination, the scrotal swelling was enormous, reaching half way down to the legs, weighing approximately 55 kilos (121 Lbs), preventing any walking motion. 
Figure 1. The scrotal swelling was enormous


He is a 5.8 feet boy, depressed at times, with crying spells. The right leg was also markedly swollen; almost two times the left leg that was spared. We were not able to palpate the external genital organs. The remainder of the physical examination was within normal limits.
A step by step surgical approach was planned, starting with the scrotal elephantiasis, aiming at removing as much affected tissue as possible, while preserving testis and penis integrity. The right leg elephantiasis repair was deferred.
Figure 2.



Surgical Procedure

The surgery took place at Christian Martinez medical Center of Jacmel, which houses the South East Urological Foundation of Haiti. A spinal anaesthesia was performed for this case. We used copious povidone for asepsis of the surgical field. We were not able, at this stage, to place a Foley catheter in the bladder, due to the fact that the penis was completely embedded in the scrotal mass. Two units of matched whole blood were transfused on the eve of the surgery and one unit during the procedure. Ceftriaxone 1 gram IV was given at incision time.
The incision started at the pedicle of the inguinal area, obliquely directed and aimed at what appeared to be the penoscrotal angle, on each side. A gelatinous material constituted the substance of the subcutaneous tissue, which was thoroughly cauterized to avoid exsanguination. Care was taken not to injure the spermatic cord and testis on both sides. Dissection was continued, alongside with hemostasis of several voluminous veins, some of them being as large as an adult patent iliac vein. 
We were truly amazed by a significant amount of arterio venous shunts discovered throughout the dissection; lymphatic tissue was overly present in the surgical field as well, but no lymph nodes were noted.
Testis and spermatic cord were ultimately identified, secured and protected outside of the surgical field. On both sides, they appeared normal at physical examination. Further dissection enabled us to identify the penis, which was catheterized with an 18 French size Foley. A third incision made below and parallel to the hypogastric transverse line, united the proximal ends of the two previous oblique incision lines. It was carried down through the dartos, with the same attention to careful haemostasis. A fourth incision on the superior aspect of the penis led to the complete degloving of the penis, which appeared to be intrinsically healthy, with  open meatus and normal corpora. At this time, a circumferential incision around the base of the scrotum, especially on the inferior aspect, was all that was needed to remove the mass, which was negatively inspected for mass lesion and then weighed. We completed the dissection by surgically cleaning the tissue margins and reinforcing haemostasis.
Then, we continued with the plastic reconstruction of the scrotum by developing skin flaps from both sides of the scrotal area. They were reconstructed towards the midline area to cover the testis and oversewn with 3-0 vicryl. The flap looked fairly healthy throughout and no tension was noted in any area of the suture line. Another flap was also developed from an inland of skin left at the dorsum of the penis and the pubic area. It was nicely isolated and wrapped around the penis, and then sutured with vicryl 3-0 at the inferior aspect of the penis. A Jackson Pratt drain was left in negative pressure in the scrotal wound. There again, no surgical tension was noted. Compress dressings were applied to the wound, putting an end to an 8 hour surgical exercise.
Figure 3.  


The post op unfolded with no particularity. Foley was removed after 8 days, leading to a normal micturition. No prophylactic heparinization was given; ceftriaxone was continued for seven days, and then switched to amoxicillin. However, an episode of wound infection did occur at 10 days post op period, warranting debridement and opening of the scrotal wound, which was then allowed to heal by secondary intention. At 6 weeks post op, the wound has completely and nicely healed off. Anti filarial management was provided, according to the protocol set forth by the Haiti Health ministry.
Figure 4. After the surgical procedure



Review of literature and discussion

Filarial elephantiasis affects about 120 million people around the world, and, in 90% of cases, is caused by Wuchereria bancrofti, a nematode transmitted by mosquito bite. The cycle of life of the nematode is straightforward, starting with the larvae inoculated to the patient through several biting events; they navigate to the blood and then move to the lymphatic system as they become adult, causing obstruction of the human lymphatic web with the help of the symbiotic endobacteria Wolbochia and an unregulated host inflammatory response.
Filarial elephantiasis is the leading cause of physical disability worldwide. The Global Alliance to Eliminate  Lymphatic Filariasis (GAELF), created in 2000 to assist in resource, advocacy and mobilization against the disease, has achieved excellent results ,with a goal of completely eliminating filariasis by 2020. Its action is well perceived in Haiti, where Wuchereria bancrofti is endemic.
Medical treatment with diethylcarbamazine and ivermectin can significantly improve the clinical outcomes in lymphatic filariasis. Surgery is used in disfiguring cases non amenable to conservative measures.

References 

1. Reconstruction of penile and scrotal lymphedema. Apesos J, Anigian G. Ann Plast Surg 1991 Dec 27(6) 510-3.
2. Giant Scrotal Elephantiasis: an idiopathic case. Dianzani C, Gaspardini F,Persichetti, P,Bruneti B,Pizzuli A, Margioti K,Degener, AM. Int J Immunopath Pharmacol 2010 Jan-Mar 23(1)369-72.
3. Surgical Management of Elephantiasis of Male Genitalia. Ollapallil JJ,Watters DA. BJU 1995 Aug 76 (2) 213-5.
4. Elephantiasis of the Penis and Scrotum. A review of 350 cases
Dandapat MC,Mohapatras K,Patro SK. AM J Surgery May 1985 149(5)686-90.
5. Penoscrotal Elephantiasis, Diagnostic and Treatment options. Zugor V,Houch R E,Labanais AP,Schneider.
6. Surgical Repair of idiopathic scrotal Elephantiasis. Zacharadis F,Duddange T, Zacharadis E, Ioannidis E. South Med  , feb 2008



Date added to bjui.org: 01/11/2010 (publication information)
This content is exclusive to bjui.org website.

Comments

Please consider his erectile function and ejaculation. In the near future, perhaps he will need rehabilitation and counseling and will be a good family man too.

9-11-2010;9.37am.

Submitted by: warawit chaianant
Date Submitted: 11/11/2010


I thank very much the reader for his comment. So far, the patient seems to have both erectile and ejacualation functions well recovered,per our last conversation a week ago. After dropping out school for the past 5 years,He is now back to school, with enhanced spirit and boosted moral. We are now planing the right leg surgery for the end of the year.

Submitted by: Jacques Jeudy
Date Submitted: 24/10/2010


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CASE REPORTS:
Surgical repair of a giant scrotal elephantiasis