Surgical Reconstruction of Giant Penoscrotal Lymphedema in Sub-Saharan Africa

Surgical Reconstruction of Giant Penoscrotal Lymphedema in Sub-Saharan Africa

Accepted Manuscript Title: Surgical Reconstruction of Giant Penoscrotal Lymphoedema in SubSaharan Africa Author: Ayo A. Salako, Jimoh K. Olabanji, Ayo...

658KB Sizes 50 Downloads 101 Views

Accepted Manuscript Title: Surgical Reconstruction of Giant Penoscrotal Lymphoedema in SubSaharan Africa Author: Ayo A. Salako, Jimoh K. Olabanji, Ayodeji O. Oladele, Gideon H. Alabi, Ifedayo E. Adejare, Rotimi A. David PII: DOI: Reference:

S0090-4295(16)30833-0 10.1016/j.urology.2016.09.064 URL 20136

To appear in:


Received date: Accepted date:

11-8-2016 10-9-2016

Please cite this article as: Ayo A. Salako, Jimoh K. Olabanji, Ayodeji O. Oladele, Gideon H. Alabi, Ifedayo E. Adejare, Rotimi A. David, Surgical Reconstruction of Giant Penoscrotal Lymphoedema in Sub-Saharan Africa, Urology (2016), 10.1016/j.urology.2016.09.064. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.

TITLE: Surgical reconstruction of giant penoscrotal lymphoedema in sub-Saharan Africa

AUTHORS: Ayo A. Salako1,4, Jimoh K. Olabanji2,4, Ayodeji O. Oladele2,4, Gideon H. Alabi3, Ifedayo E. Adejare3, Rotimi A. David1

AUTHOR AFFILIATIONS: 1- Urology Unit, Department of Surgery, Obafemi Awolowo University Teaching Hospitals Complex, Ile-Ife, Nigeria 2- Plastic surgery unit, Department of Surgery, Obafemi Awolowo University Teaching Hospitals Complex, Ile-Ife, Nigeria 3- Department of Surgery, Federal Medical Centre, Owo, Ondo State, Nigeria 4- Department of Surgery, Obafemi Awolowo University, Ile-Ife, Nigeria

CORRESPONDING AUTHOR: Prof A.A Salako, Department of Surgery, Obafemi Awolowo University, Ile-Ife, Nigeria Tel: +2348033455165 Email: [email protected]


Page 1 of 19

WORD COUNT Abstract: 188 Manuscript: 2,076

KEYWORDS: Surgical reconstruction, filariasis, giant scrotum, lymphoedema, SubSaharan Africa.


FUNDING: No extra funding was received for this study

ACKNOWLEDGEMENT: We thank the surgical support staff in urology and plastic surgery units of our hospital, who were involved in the management of the patients. ABSTRACT Objective: To present management challenges, surgical technique and outcome associated with penoscrotal reconstruction in patients with giant scrotal lymphoedema in sub-Saharan Africa.

Method: A prospective study of all patients who had penoscrotal reconstruction for giant scrotal lymphoedema at our university teaching hospital between January 2003 and December 2012 was carried out. Patients’ pre-operative clinical evaluation findings, operative technique and post-operative course were reviewed after obtaining ethical approval and informed consent from the patients. 2

Page 2 of 19

Results: Nineteen patients presented to us with giant scrotal lymphoedema during the period of study out of which eleven had surgical excision and were studied. Their mean age and median duration of symptoms were 48.5 years and 11.5 years respectively. They all had surgical reconstruction using modified Charles procedure by the same combined team of urologists and plastic surgeons. Scrotal hematoma (27.3%) and superficial surgical site infection (18.2%) were complications encountered postoperatively. One patient (9.1%) had recurrence within 24 months, requiring repeat excision.

Conclusion: Giant scrotal lymphoedema poses severe physical challenge to the sufferer. Surgery remains the only hope to reduce penoscrotal size. Combined effort of urologic and plastic surgeons is essential for reconstruction.


Scrotal lymphoedema is an uncommon condition in surgical practice worldwide. It results from obstruction, aplasia or hypoplasia of lymphatic vessels at the inguinal region with lymph stasis in the scrotum and subsequent fibrous hyperplasia and hypertrophy, in a chronic inflammatory process1,2. The etiology varies, but most cases result from filarial infestation, especially in tropical African countries. Chronic 3

Page 3 of 19

granulomatous conditions, groin surgery, irradiation and malignancy are less common causes, and are more important in areas where filariasis is non-endemic3,4. More rarely, it may be idiopathic or congenital, often occurring in association with lymphoedema of the limbs5. The condition is usually progressive, sometimes attaining giant sizes (often larger than the patient’s head) if no timely intervention is offered. In addition to the grotesque appearance, progression to a giant size impairs ambulation, makes sexual intercourse impossible, prevents voiding in the standing position and mitigates against proper hygiene of the perineal region and external genitalia, leading to mal-odor and recurrent episodes of skin infection, thereby adversely affecting the patient's quality of life and self-esteem1,2. Although medical therapy such as the use of filaricides result in rapid elimination of the filarial worms, the lymphoedematous changes they cause is often recalcitrant to medical therapy. Other medical treatment options such as the use of diuretics and scrotal elevation are equally not effective and are no longer recommended6,7. Pressure therapy, though effective in the management of limb lymphoedema, is not appropriate for scrotal lymphoedema. Surgery therefore offers the only hope of achieving appropriate reduction in the size of the tissue, and this can sometimes be challenging8. Using the cases of giant scrotal lymphoedema managed in our hospital in sub-Saharan Africa over a decade, we highlight this uncommon condition of giant scrotal lymphoedema. We discuss the details of surgical penoscrotal reconstruction, as well as the challenges and outcome of managing the giant scrotum.


Page 4 of 19


All patients who had surgical reconstruction for penoscrotal lymphoedema between January 2003 and December 2012 were recruited into the study after informed consent and ethical approval from the research and ethics committee of our university teaching hospital. Same combined team of reconstructive urologists and plastic surgeons managed all of the patients. Under appropriate anesthesia, the patients were positioned supine and prophylactic intravenous Amoxicillin-clavulanic acid antibiotic administered. Urethral catheter was passed for urine drainage. One of them, who had co-existing urethral stricture with “watering-can perineum” from multiple perineal urethrocutaneous fistulae had suprapubic cystostomy for proximal urine diversion in place of urethral catheterization. The operation was commenced by making an incision over the external inguinal ring bilaterally and deepening it to access the cords which were mobilized and delivered into the wound together with the testes. The incision was extended over the scrotum and generous excision of all grossly affected penoscrotal skin and subcutaneous tissues down to the Buck’s fascia was done, in a modified Charles procedure. The resultant defect on the penile skin was covered by rolling back the uninvolved preputial skin along the penile shaft while a new scrotum was fashioned from the apron of normal skin at the scrotal neck that had been stretched by the mass. The tunica vaginalis was everted


Page 5 of 19

around the testes before placement in the neoscrotum after which the wound was closed over a drain and scrotal support/elevation done with crepe bandage. Postoperative care involved regular wound dressings, antibiotics, analgesics and other routine treatment as appropriate. The drain was removed when its output was less than 20mls/day of serous effluent, usually at about the 4th post-operative day. The patient with the co-existing urethral stricture later had urethroplasty with good outcome. Clinical data regarding the age, duration of symptoms, surgical reconstruction technique, length of hospital stay, post-operative complication, duration of follow up and treatment outcome were entered into a proforma designed for the study and analyzed using the statistical package for social sciences version 21.


A total of 19 patients with giant scrotal lympoedema were seen during the period out of which 11 (57.9%) had surgical intervention. Their ages ranged between 23 and 63 years (mean 48.5 years) while the median duration of symptoms was 11.5 years. The length of hospital stay was between 20 days and 43days with mean of 26.3days. None of the patients had microfilaria in the blood stream. Average weight of excised specimen was 22.4kg while the mean follow up period was 36.5 months. Post-operatively, three patients (27.3%) had scrotal hematoma managed with scrotal support and elevation, 6

Page 6 of 19

two (18.2%) had surgical site infection which responded to topical dressing and antibiotics while one (9.1%) had disease recurrence after 24 months of follow-up, necessitating repeat excision and scrotal reconstruction. Further clinical and perioperative information of the patients are presented in table 1 while the pre- and postoperative clinical photographs of one of the patients are highlighted in Figure 1-5.


This study reviewed the surgical technique as well as management challenges of giant penoscrotal lymphoedema in a typical sub-Saharan African setting. The giant variants of penoscrotal lymphoedema


potential causes of

physical disability,


embarrassment and psychological disturbance for affected patients2. Though lymphoedema is relatively common in the tropics due to the endemic nature of filariasis from Wuchereria bancrofti infestation9, giant penoscrotal lymphoedema is not so common10. Surgical intervention remains the mainstay of management since nonsurgical approaches are ineffective2,6. In spite of this, only 11 (57.9%) of patients that presented to us with giant penoscrotal lymphoedema had surgery while others defaulted, possibly due to stronger belief in traditional herbal medicine or secondary to financial constraint, since health insurance schemes are not yet fully established in our domain. These may also explain the delayed hospital presentation of our patients evidenced by the relatively long symptom duration (median 11.5 years) before 7

Page 7 of 19

presentation. One of the patients, with co-existing urethral stricture, already had “watering-can








presentation. This further buttresses the fact that patients generally present late in our environment, and many actually relate to orthodox hospitals only as a last resort. The mean age of the patients studied (48.5 years) is within similar age range reported from other studies2,11. None of our patients had microfilaria in the blood film at presentation. This may be explained by the use of anti-filarial medications distributed free in many communities as part of eradication programmes for filariasis. A report from a similar setting as ours also found no evidence of filariasis in their index patient with giant penoscrotal lymphoedema2. Such scenario may however constitute a diagnostic challenge in determining the underlying aetiology of giant penoscrotal lymphoedema. Our unit protocol for such diagnostic dilemma is to empirically assume the lymphoedema was caused by background chronic filariasis if the patient has no other obvious risk factors for scrotal lymphoedema and is resident in a tropical region known to be endemic for the disease. All our patients had modified Charles procedure, with preservation of the testes and spermatic cords and use of local skin flaps for wound closure. Similar treatment has been successfully used for patients with massive penoscrotal lymphoedema by other authors2,3,7,11. Lymphangioplasty or microlymphatico-venous anastomosis was not feasible in any of our patients due to obliteration of appropriate lymphatic channels from chronic fibrosis secondary to late presentation of cases. The average weight of the excised specimen from our study (22.4kg) is within range of 8.5-30kg reported in world literature10,12. Although extensive scrotal skin loss due to 8

Page 8 of 19

trauma or infection may require coverage of the testes with pedicled fascio-cutaneous flaps, this is usually unnecessary in the presence of scrotal lymphoedema as the stretched adjoining apron of normal skin is usually available for refashioning a new scrotum13. We found this to be very useful in our practice. Other authors have also found its use to be adequate and comparable to other techniques in providing a scrotal appearance that is close to normal7,14. Because vaginal hydrocele is a relatively common co-existing condition with scrotal lymphoedema (due to lymphatic obstruction and/or host response to adult worms15), we routinely drained all hydroceles and everted the tunica vaginalis while placing the testes in the neoscrotum created. A major challenge reported by other authors during the procedure is the reconstruction of the penile skin. Options such as local flaps or split thickness skin grafts have been used successfully in literature2,7,11,16. We were able to achieve closure of the penile skin defect by an adaptation involving rolling back of the unaffected preputial skin along the penile shaft. Singh et al11 also used similar modification with success. Post-operative scrotal hematoma encountered in our study (27.3%) is not unusual, due to dependent nature of the scrotum and the fact that scrotal reconstruction has higher morbidity and risk of complications8. Use of wound drain and scrotal support in the postoperative period helped to limit its occurrence. Surgical site infection (18.2%) may have been caused by poor hygiene in some of our patients, some of whom also apply local concoctions to soil the wound site after discharge from the hospital. A similar study by Singh et al11 recorded surgical site infection in 25% of patients in their series. 9

Page 9 of 19

The recurrence experienced in one patient (9.1%) was quite surprising as all the grossly diseased penoscrotal tissue was excised at first surgery and he did not have any obvious risk factors for recurrence such as groin surgery or radiation exposure. He has however had a repeat excision with good results. All our patients could be said to have had an overall satisfactory outcome from reconstructive surgery as they were able to ambulate better and void in the standing position after the operation. In addition, they were satisfied with the subjective final cosmetic appearance of their external genitalia after the surgery.


Penoscrotal reconstruction is a safe and effective management option for giant penoscrotal lymphoedema in resource-limited sub-Saharan African settings. Combined effort of reconstructive urologists and plastic surgeons is essential for good outcome.


Modolin M, Mitre AI, Da Silva JC, Cintra W, Quagliano AP, Arap S et al. Surgical treatment of lymphedema of the penis and scrotum. Clinics (Sao Paulo, Brazil). 2006;61(4):289-294


Rahman GA, Adigun IA, Yusuf IF, Aderibigbe AB, Etonyeaku AC. Giant scrotal lymphedema of unclear etiology: a case report. Journal of medical case reports. 2009;3:7295-7299 10

Page 10 of 19


Kuepper D. Giant scrotal elephantiasis. Urology. 2005;65(2):389 e319 - e321


Murphy MJ, Kogan B, Carlson JA. Granulomatous lymphangitis of the scrotum and penis. Report of a case and review of the literature of genital swelling with sarcoidal granulomatous inflammation. Journal of cutaneous pathology. 2001;28(8):419-424


Wananukul S, Jittitaworn S. Primary congenital lymphedema involving all limbs and genitalia. Journal of the Medical Association of Thailand. 2005;88(12):19581961


Kumar P, Navaneethan GP. Resection of scrotal lymphedema. Plastic and reconstructive surgery. 2005;116(2):24e-27e


Dandapat MC, Mohapatro SK, Patro SK. Elephantiasis of the penis and scrotum: A review of 350 cases. The American journal of surgery. 1985;149(5):686-690


Lim KHA, Speare R, Thomas G, Graves P. Surgical Treatment of Genital Manifestations of Lymphatic Filariasis: A Systematic Review. World Journal of Surgery. 2015;39(12):2885-2899


Shenoy R, Sandhya K, Suma T, Kumaraswami V. A preliminary study of filariasis related acute adenolymphangitis with special reference to precipitating factors and treatment modalities. Southeast Asian Journal of Tropical Medicine and Public Health. 1995;26:301-305


Parmar HD. The surgical approach in huge scrotal lymphedema. Int J Med Sci Public Health. 2013;2(1):153-155


Singh V, Sinha RJ, Sankhwar SN, Kumar V. Reconstructive surgery for penoscrotal filarial lymphedema: a decade of experience and follow-up. Urology. 2011;77(5):1228-1231


Van Der Walt JC, Perks TJ, Zeeman BJ, Bruce-Chwatt AJ, Graewe FR. Modified Charles procedure using negative pressure dressings for primary lymphedema: a functional assessment. Annals of plastic surgery. 2009;62(6):669-675


Kochakarn W, Hotrapawanond P. Scrotal reconstruction using thigh pedicle flaps: long-term follow-up of 12 cases. Journal of the Medical Association of Thailand. 2001;84(12):1738-1742


Ottesen EA. Editorial: The global programme to eliminate lymphatic filariasis. Tropical Medicine & International Health. 2000;5(9):591-594 11

Page 11 of 19


Hussein O, El Setouhy M, Ahmed ES, Kandil AM, Ramzy RM, Helmy H et al. Duplex Doppler sonographic assessment of the effects of diethylcarbamazine and albendazole therapy on adult filarial worms and adjacent host tissues in Bancroftian filariasis. The American journal of tropical medicine and hygiene. 2004;71(4):471-477


Sun GC, Zhong AG, He W, Du P, Song WM, Ma JG. Reconstruction of the external genitals and repair of skin defects of the perineal region using three types of lateral groin flap. Annals of plastic surgery. 1990;24(4):328-334



Figure 1:

Pre-operative image for one of our patients (anterior view)


Figure 2:

Pre-operative image for same patient in Figure 1 (posterior view)


Figure 3:

Post-operative image taken at 6th month of follow-up for patient in Figure 1


Figure 4:

Immediate post-operative image of patient in figure 1, showing skin closure with local flaps


Figure 5:

Follow-up at 6 months in same patient shown in figure 1


Page 12 of 19





Operative findings

Length of

Post operative

hospital stay



(Clavien classification) 1


Thickened scrotal skin, edematous penis, normal










testes and spermatic cord 2


Verrucus scrotal skin, buried penis, left testicular atrophy, normal right testis and spermatic cord



Thickened scrotal skin, buried penis,


testes, normal spermatic cord 4


hematoma (II)

Hyperpigmented and thickened scrotal skin,

24 days

buried penis, normal testes and spermatic cord 5


Hyperkeratotic scrotal skin, swollen penis, normal



(IIIa) 24 days

testes and spermatic cord 6




hematoma (II)

Dyschromic changes on scrotal skin, partially

43 days




Page 13 of 19

buried penis, normal testes and spermatic cord, co-existing urethral stricture with “watering-can” perineum 7


Thickened scrotal skin, swollen penis, normal

30 days



21 days



28 days



testes and spermatic cord 8


Hyperkeratotic scrotal skin, partially buried penis, normal testes and spermatic cord



Hyperkeratotic scrotal skin, swollen penis, normal testes and spermatic cord



hematoma (II)

Hyperkeratotic scrotal skin, buried penis, normal

22 days



24 days



testes and spermatic cord 11


Hyperkeratotic scrotal skin, swollen penis, normal testes and spermatic cord

Satisfactory outcome implies grossly reduced penoscrotal size post-operatively with improved ambulation, ability to void in the standing position and a subjective cosmetically acceptable final outcome SSI: Surgical site infection 14

Page 14 of 19

* The patient had recurrence after 24 months ** Repeat excision and reconstruction done with good results


Page 15 of 19

I have no conflicts or disclosures relevant to this editorial comment. Kenneth W. Angermeier, MD


Page 16 of 19

Title: Editorial Comment on Manuscripts URL-D-16-01644R1 and URL-D-16-01665R1

Kenneth W. Angermeier, MD, FACS Professor of Surgery Director, Center for Genitourinary Reconstruction Glickman Urological and Kidney Institute Cleveland Clinic Cleveland, OH

Ph: 216-444-0415 Email: [email protected]


Page 17 of 19

Marked genital lymphedema, although relatively uncommon, has been a well-recognized entity for many years1. This condition has a significant impact on quality of life with regard to voiding and sexual function, ambulation and hygiene. Over time there has been little change in the method of treatment, which consists of surgical excision of the affected skin and subcutaneous tissues with subsequent genital reconstruction using local flaps and skin grafts. The study by Salako, et al describes 19 patients with giant penoscrotal lymphedema, 11 of whom underwent surgery. Etiology was presumably related to chronic filariasis in most cases, although none had microfilaria identified in the blood film at presentation. Following excision, penile shaft coverage was accomplished with use of the uninvolved inner preputial skin. In our experience, the amount of this preputial skin has been limited and often insufficient for coverage of the entire penile shaft, but it is certainly something to keep in mind in these difficult cases. Outcome was satisfactory in all cases with improved voiding and ambulation, however one patient developed recurrence 2 years later requiring repeat surgery. Wisenbaugh, et al report on the surgical treatment of 11 patients with massive localized lymphedema (MLL) of the scrotum. MLL was described as a clinical entity in 1998, and was characterized as localized edema in the extremities of obese patients in order to help distinguish it from well differentiated liposarcoma. In that study, the authors state that it has a morphologic identity to diffuse lymphedema2. This clinical diagnosis has since been extrapolated to describe scrotal lymphedema in obese patients, however this does not seem to be a new entity, just a change in nomenclature. Most of the patients with genital lymphedema that we have seen are also significantly obese. A few of these patients have developed lymphedema below a prior panniculectomy scar, which may contribute to the condition as well. Regardless of the label, the treatment remains the same. In agreement with the authors, we try to avoid a skin graft to the penis if possible. The chronically stretched skin extending from the pubic region to the mass of lymphedema in some cases may be fashioned into a flap to cover the penile shaft with a very good functional outcome 3. The findings with regard to weight gain after the procedure are well documented by the authors and appropriate. We have also treated patients who were emphatic that the genital lymphedema was limiting their ability to exercise and lose weight, only to have them return after successful surgery weighing the same or more. In summary, genital lymphedema is a condition that will continue to be encountered by reconstructive surgeons around the world. As demonstrated by the authors, appropriate preoperative counseling, careful surgical technique, and meticulous postoperative wound care will result in an improved appearance and quality of life in the majority of patients. References 18

Page 18 of 19

1. Garaffa G, Nim C, Ralph DJ. The management of genital lymphedema. BJUI. 2008;102:480484. 2. Farshid G, Weiss SW. Massive localized lymphedema in the morbidly obese: a histologically distinct reactive lesion simulating liposarcoma. Am J Surg Pathol. 1998;22:1277-1283. 3. Higuchi TT, Yamaguchi Y, Wood HM, Angermeier KW. Evaluation and treatment of adult buried penis. Curr Urol Rep. 2012;13:277-284.


Page 19 of 19