Surgical Management of Massive Scrotal Lymphedema: A Case Report and Review of the Literature

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Abstract Scrotal lymphedema, or scrotal elephantiasis, is a rare progressive condition caused by impaired lymphatic drainage and accumulation of protein-rich interstitial fluid, resulting in fibrosis and massive genital enlargement. This case report presents a 52-year-old man with massive idiopathic scrotal lymphedema, or scrotal elephantiasis, characterized by a 10-year history of progressive scrotal enlargement, recurrent infections, and loss of urinary and sexual function with a completely buried penis Diagnostic evaluation excluded infectious, neoplastic, and vascular causes, confirming non-filarial scrotal lymphedema. Management consisted of staged radical excision of fibrotic tissue with preservation of the testes, spermatic cords, and phallus, followed by reconstruction using a pedicled superficial circumflex iliac artery perforator groin flap. Adjunctive Z-plasty at the penile base and full-thickness thigh skin grafts restored contour and mobility. Postoperative recovery was uneventful, with complete flap survival and restoration of urinary function, hygiene, mobility, and quality of life. This case demonstrates that radical excision with SCIP flap reconstruction provides a reliable and durable solution for massive idiopathic scrotal lymphedema, including cases requiring tissue resection exceeding 30 kg. A focused review of the literature places this case in the context of previously reported cases of massive scrotal lymphedema.
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Surgical Management of Massive Scrotal Lymphedema: A Case Report and Review of the Literature | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case Report Surgical Management of Massive Scrotal Lymphedema: A Case Report and Review of the Literature Alexander H. Chang, Natalia Mejia, Aryan Gupta, Jing Qin Tay, and 5 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-8652655/v1 This work is licensed under a CC BY 4.0 License Status: Under Revision Version 1 posted 10 You are reading this latest preprint version Abstract Scrotal lymphedema, or scrotal elephantiasis, is a rare progressive condition caused by impaired lymphatic drainage and accumulation of protein-rich interstitial fluid, resulting in fibrosis and massive genital enlargement. This case report presents a 52-year-old man with massive idiopathic scrotal lymphedema, or scrotal elephantiasis, characterized by a 10-year history of progressive scrotal enlargement, recurrent infections, and loss of urinary and sexual function with a completely buried penis Diagnostic evaluation excluded infectious, neoplastic, and vascular causes, confirming non-filarial scrotal lymphedema. Management consisted of staged radical excision of fibrotic tissue with preservation of the testes, spermatic cords, and phallus, followed by reconstruction using a pedicled superficial circumflex iliac artery perforator groin flap. Adjunctive Z-plasty at the penile base and full-thickness thigh skin grafts restored contour and mobility. Postoperative recovery was uneventful, with complete flap survival and restoration of urinary function, hygiene, mobility, and quality of life. This case demonstrates that radical excision with SCIP flap reconstruction provides a reliable and durable solution for massive idiopathic scrotal lymphedema, including cases requiring tissue resection exceeding 30 kg. A focused review of the literature places this case in the context of previously reported cases of massive scrotal lymphedema. Figures Figure 1 Figure 2 Figure 3 Figure 4 Figure 5 Figure 6 Introduction Lymphedema is a chronic, progressive disorder characterized by impaired lymphatic drainage and accumulation of protein-rich interstitial fluid within soft tissues, leading to inflammation, fibrosis, and progressive hypertrophy of the affected region. 1 Recurrent lymphatic obstruction triggers chronic inflammation, fibroblast proliferation, and adipose deposition, resulting in irreversible tissue thickening and deformity. 2 Although most commonly involving the extremities, genital lymphedema can cause profound functional disability and psychosocial distress. 3 Lymphedema involving the genitalia can be classified as primary, due to congenital lymphatic dysplasia, or secondary, arising from infection, malignancy, trauma, obesity, or previous surgical or radiation injury. 4,5 In endemic regions such as sub-Saharan Africa, Southeast Asia, and parts of South America, filarial infection remains the leading cause of genital lymphedema. 2,3 In nonendemic areas, chronic inflammatory, neoplastic, or iatrogenic mechanisms predominate. 5 Progressive scrotal involvement may result in massive enlargement, recurrent infection, and significant impairment of mobility, hygiene, urination, and sexual activity. In severe cases, the mass effect can become debilitating, causing restriction in daily activities and psychosocial distress. 1 The underlying tissue changes include fibrosis, dermal thickening, and lymphangiectasia, which render conservative measures such as compression, massage, and antibiotics largely ineffective once fibrosis becomes established. 5,6 Surgical excision of diseased tissue with preservation of viable structures remains the definitive treatment for chronic or massive lymphedema. 7,8 The primary goals of surgery are to restore penile and scrotal anatomy, reduce infection risk, and re-establish urinary and sexual function. 4 Successful outcomes depend on accurate preoperative mapping, complete removal of fibrotic tissue, and tension-free reconstruction that provides thin, pliable coverage while minimizing donor site morbidity. 1,5 Multiple surgical techniques have been described for genital lymphedema, and the choice of approach depends on disease severity, tissue viability, and reconstructive goals. Local excision with primary closure may suffice for limited disease, whereas advanced cases often require split-thickness skin grafting or flap coverage to achieve durable, pliable reconstruction. 3,9 Flaps such as the gracilis, anterolateral thigh (ALT), and superficial circumflex iliac artery (SCIA) or superficial circumflex iliac artery perforator (SCIP) flaps provide well-vascularized tissue suitable for resurfacing and functional restoration. 10 This case report describes the management of massive scrotal lymphedema complicated by a buried penis and severe reduction in urinary and sexual function. The magnitude of disease required comprehensive diagnostic evaluation to exclude infectious, neoplastic, and vascular causes and to delineate the extent of fibrotic and viable tissue. Precise preoperative mapping was critical to identify preserved penile and testicular structures, guide resection margins, and anticipate the reconstructive requirements. Case A 52-year-old man from a tropical region presented with a 10-year history of progressive scrotal enlargement. He reported increasing difficulty with activities of daily living. Walking and sitting were particularly challenging, as the scrotum extended to his lower legs and rested on the floor when he stood upright (Fig. 1 ). The mass had become ulcerated, placing him at risk for systemic infection. The scrotum weighed approximately 30 kg and completely buried the penis within the hypertrophic folds, resulting in multiple urinary tract infections over the years (Fig. 2 ). Chronic inflammation and recurrent infection led to urethral strictures, ultimately preventing complete evacuation of urine. Despite severe physical and psychological distress, the patient had not sought medical attention earlier due to financial limitations and poor access to definitive surgical treatment. The patient had no significant past medical history. On examination, the scrotum was grossly enlarged, extending to the level of the calves, with thickened, indurated skin consistent with elephantiasis. Physical examination also revealed a buried penis with loss of sexual function. The presence of multiple urethral strictures, a collapsed urethral lumen, and massive scrotal bulk precluded urinary evacuation. Filariasis was excluded early in the diagnostic process through negative peripheral smear results and the absence of systemic manifestations. Multiple punch biopsies revealed nonspecific chronic inflammation and dermal fibrosis without evidence of malignancy or infection. Ultrasound and CT imaging ruled out underlying masses. Pelvic CT demonstrated markedly dilated lymphatic channels and diffuse thickening of the scrotal tissue, confirming the diagnosis of idiopathic non-filarial scrotal lymphedema (Fig. 3 ). CT angiography showed no significant arterial or venous compromise. Given the profound anatomic distortion and total loss of function, surgical intervention was deemed necessary. A multidisciplinary team proceeded with a two-stage surgical approach. The first stage focused on debulking and excision of redundant scrotal tissue to expose the penis and testes, followed by definitive reconstruction. A horizontal incision was made in the groin area, with the anatomical outline premarked to map the skin for future defect coverage. The size and extent of the incision were determined by the anticipated defect. Careful undermining of the abdominal wall was performed to preserve perforating arteries within the abdominal tissue. Skin dissection was carried out to facilitate wound approximation following excision of the excessive scrotal tissue. Dissection continued distally to the pubis, with the goal of identifying and assessing the phallus. At this stage, the urology team assumed operative management for tissue salvage and revision of urethral strictures and testicular anatomy. Urethroscopy was performed for stricture release, followed by placement of a permanent Foley catheter. The testes and spermatic cords were inspected and found to be anatomically and functionally preserved (Fig. 4 ). The second and final stage was performed by the plastic surgery team. A meticulous perineal dissection was carried out, with emphasis on identifying the anterior and posterior tissue boundaries to guide bilateral flap design. A superficial circumflex iliac artery perforator (SCIP) flap was selected to cover the genitals and protect the exposed spermatic cords. The anterior superior iliac spine and pubic tubercle served as key landmarks during flap harvest. After incising the deep fascia, subfascial dissection was performed to identify the superficial circumflex iliac artery, which was traced to its origin from the femoral artery, allowing mobilization of the pedicle for posterior rotation of the flap. The deeper perineal layers were closed using absorbable monofilament sutures, while the superficial dermal layer was closed with Vicryl. Multiple drains were placed for lymphatic drainage and fluid management. To optimize functional outcomes, a Z-plasty was performed at the penile base following release, and full-thickness skin grafts harvested from the inner thigh were used for shaft reconstruction (Fig. 5 ). Postoperative recovery was uneventful. No wound dehiscence or revision was required. The patient was able to void normally after reconstruction. At a 3-month follow-up, the patient reported marked improvement in mobility, hygiene, and quality of life. Discussion Scrotal lymphedema is a rare and debilitating condition characterized by chronic lymphatic obstruction leading to progressive enlargement and fibrosis of the scrotal tissues. 1 Although most cases in tropical regions are caused by filarial infection, this patient’s presentation was non-filarial, supported by negative parasitological and serologic testing. 2 Non-filarial genital lymphedema may arise secondary to neoplastic, inflammatory, or idiopathic causes. 5 The chronicity of this case and absence of secondary pathology support a diagnosis of idiopathic non-filarial scrotal lymphedema. The earliest detailed description of surgical management dates to 1820, when Professor Jacques-Mathieu Delpech of Montpellier performed radical excision of a 60-pound scrotal mass in Jean Baptiste Authier, a cavalryman disabled by syphilitic lymphatic obstruction. 11 The operation was carried out without anesthesia before an audience of 300, establishing the enduring principles of complete resection and local flap reconstruction later depicted in Delpech’s operative drawings. 11 Subsequent surgeons, including Matas (1913), Young (1926), and DeSavitsch (1941) refined these methods while maintaining the same operative philosophy. Together, these historical precedents illustrate that despite technical advances over two centuries, the fundamental principle has remained unchanged: radical excision of diseased tissue with tension-free, well-vascularized coverage remains the cornerstone of treatment for genital elephantiasis. To contextualize our case within the broader literature, a comprehensive review was performed. A total of 350 articles were identified, of which 60 met inclusion criteria for studies addressing surgical management of scrotal lymphedema. These included 31 case reports, 25 case series, and 4 literature reviews published between 1977 and 2025. The screening and selection process is summarized in Fig. 6 . Our case presentation of a 32 kg resection ranks among the largest documented in the last decades, with reported resection weights across the literature ranging from 0.9 to 61 kg. Representative modern series are summarized in Table 1 . Wisenbaugh et al. described the heaviest excision to date (61 kg) managed by wide en bloc resection and primary closure using preserved lateral and posterior scrotal flaps. 12,13 Tekin et al. describes surgical management of a 42 kg idiopathic case through subtotal scrotectomy and posterior flap advancement, avoiding grafting entirely. Haq et al. 14 reported a 33 kg resection managed with vacuum-assisted closure and delayed split-thickness grafting, while Welling et al. describes staged debulking and resurfacing in a morbidly obese patient with a BMI of 77, achieving full functional recovery. 15 Salako et al. used a modified Charles procedure for a 22.4 kg case, employing preserved preputial and scrotal-neck skin for primary closure. 16 Table 1 Reported cases of massive scrotal or penoscrotal lymphedema requiring large-volume excision, including operative technique and resection weight. Note * was the reported average, not the maximum singular weight. Across these large resections, outcomes consistently favored complete excision with primary closure using viable local tissue over graft-only repairs, with lower complication rates and superior functional results. When native skin was insufficient, split-thickness skin grafts (STSG) provided temporary coverage but were prone to contracture and breakdown. Consequently, regional and pedicled flaps, most commonly based on the superficial circumflex iliac artery (SCIA), anterolateral thigh (ALT), and gracilis flaps, became the preferred reconstructive options, offering thin, vascularized coverage and reduced recurrence as summarized in Table 2 . Author (Year) Resection Technique Weight of Resection (kg) Wisenbaugh et al., 2018 En bloc excision of massive, localized lymphedema with preservation of viable lateral and posterior scrotal flaps for primary closure, avoiding grafts when possible 61 Tekin et al., 2021 Subtotal scrotectomy performed emergently for hemorrhagic idiopathic scrotal elephantiasis; excision extended through lateral folds to posterior wall 42 Thejeswi et al., 2012 Near-total excision of scrotal skin; circumferential penile dissection; debulking using cautery and Ligasure; bilateral orchiectomy due to atrophy 32 Welling et al., 2022 Two-stage excision and reconstruction in a morbidly obese (BMI 77) patient; initial wide debulking followed by delayed penile resurfacing 26 Salako et al., 2018 Modified Charles procedure (i.e., complete excision of all diseased penoscrotal tissue to Buck’s fascia with immediate reconstruction using preserved healthy preputial and scrotal-neck skin for primary closure over a drain) 22.4* Haq et al., 2023 Staged excision of giant penoscrotal lymphedema using vacuum-assisted closure (VAC) to control edema and prepare wound bed for delayed grafting 20 Table 2 Table highlighting reconstruction techniques of largest reported mass resections. Author (Year) Penile Reconstruction Scrotal Reconstruction Closure Wisenbaugh et al., 2018 Native penile skin preserved; grafting deferred to minimize breakdown risk Lateral and posterior flaps advanced medially to reconstitute scrotum Primary closure with drains Tekin et al., 2021 Buried penis reconstructed using residual preputial skin; STSG considered but not required Posterior scrotal wall advanced and folded medially to create neoscrotum Primary closure without drains Thejeswi et al., 2012 Bilateral scrotal skin flaps advanced with Y-shaped midline closure Native penile skin preserved; no grafts or distant flaps used Primary closure using preserved Y-shaped scrotal flaps Welling et al., 2022 Penile shaft left degloved initially, resurfaced secondarily with STSG Lateral scrotal flaps preserved and advanced medially to form neoscrotum Two-stage closure Salako et al., 2018 Uninvolved preputial skin rolled back along shaft for penile resurfacing Neoscrotum fashioned from stretched healthy neck skin; tunica vaginalis everted around testes Primary closure with drain; scrotal support post-op Haq et al., 2023 Split-thickness skin graft (STSG) applied after edema resolution Local medial and posterior skin flaps preserved for coverage and contouring Delayed closure following VAC therapy The SCIP flap used in our case represents the modern evolution of the classical McGregor groin flap. McGregor and Jackson originally described a pedicled groin flap based on the SCIA and SIEA systems for upper extremity coverage, and their seminal paper highlighted its reliability but also noted its bulk and limited contour precision. 17 Subsequent refinements of the SCIA system, including the development of perforator-based SCIA and SCIP designs, have produced thin, pliable, and well-vascularized skin with minimal donor-site morbidity. These advances make contemporary SCIA-based flaps particularly well suited for genital and perineal resurfacing. While the SCIA and SCIP flaps share a vascular origin, they serve different reconstructive goals. The SCIA flap, derived from the classical groin or McGregor flap, provides a reliable pedicled option with thicker and more robust fasciocutaneous tissue suitable for larger perineal, suprapubic, or inguinal defects and for cases where microsurgery is not feasible or the wound bed is compromised. In contrast, the SCIP flap offers thin, pliable tissue with minimal donor-site morbidity, making it well suited for genital, perineal, and distal extremity resurfacing where contour and precision are critical. In our case, the extent and location of the defect necessitated a thin flap transfer, for which the SCIP flap provided superior functional and aesthetic outcomes. Across the modern literature, outcomes following SCIA-based reconstruction are consistently favorable, with most patients achieving restored urinary function, improved hygiene, and enhanced mobility within three to six months, and only minor wound dehiscence or seroma formation reported. As perforator and lymphatic-restorative techniques continue to evolve, the SCIA and SCIP flaps remain among the most reliable and reproducible regional options for definitive reconstruction after massive genital lymphedema. Although disruption of lymphatic drainage has long been recognized, with early attempts including permanent suturing of the scrotum to the thigh to facilitate alternative drainage pathways, reconstructive options at the time lacked the technical means to reliably address this problem. 11 With the advancement of microsurgical techniques, newer options have expanded the reconstructive armamentarium. Perforator flaps as well as lymphatic-restorative flaps vascularized lymph node transfers, provide more physiologic coverage while potentially reestablishing lymphatic drainage. 10,18,19 These innovations reflect a paradigm shift from purely excisional to functionally restorative reconstruction, particularly in centers with microsurgical capability. Our case resonates with the literature in that wide excision combined with vascularized regional tissue coverage remains the cornerstone of management. Microsurgical and lymphatic-restorative techniques represent promising advances; however, access to these treatments remains limited, particularly in low- and middle-income countries (LMICs), and long-term outcome data are sparse. The pedicled SCIA flap and its descendant the SCIP remain the most versatile, reproducible, and globally applicable options for definitive reconstruction following massive scrotal lymphedema. Declarations Ethics approval and consent to participate: All ethics protocol for institutions were observed; patient’s consent for images to be used for publication is documented. Clinical trial number Not applicable Consent for publication: Photographs were taken with consent; written informed consent for publication of their clinical details and/or clinical images was obtained from the patient. A copy of the consent form is available for review by the Editor of this journal. Competing interests: The author declares no competing interests. Acknowledgements Not applicable. Funding: None. Author Contribution Alexander H. Chang, Natalia Mejia, Aryan Gupta, Jing Qin Tay, Ambroise Gilles, Sthefano Araya, Adam C. Walchak, Sameer A. Patel, Andres Romero have all contributed equally to the manuscript. Data Availability Not applicable. References McDougal WS. Lymphedema of the External Genitalia. J Urol . 2003;170(3):711–716. doi:10.1097/01.ju.0000067625.45000.9e Dandapat MC, Mohapatro SK, Patro SK. Elephantiasis of the Penis and Scrotum. A Review of 350 Cases. Am J Surg . 1985;149(5):686–690. doi:10.1016/s0002-9610(85)80156-2 Kaciulyte J, Garutti L, Spadoni D, et al. Genital Lymphedema and How to Deal with It: Pearls and Pitfalls from over 38 Years of Experience with Unusual Lymphatic System Impairment. Medicina (Mex) . 2021;57(11):1175. doi:10.3390/medicina57111175 Holman CM, Arnold PG, Jurkiewicz MJ, Walton KN. Reconstruction of Male External Genitalia with Elephantiasis. Urology . 1977;10(6):576–578. doi:10.1016/0090-4295(77)90108-X Brewer MB, Singh DP. Massive Localized Lymphedema: Review of an Emerging Problem and Report of a Complex Case in the Mons Pubis. Ann Plast Surg . 2012;68(1):101–104. doi:10.1097/SAP.0b013e318211910e Bogusz AM, Hussey SM, Kapur P, Yan Peng, Tunc Gokaslan S. Massive Localized Lymphedema With Unusual Presentations: Report of 2 Cases and Review of the Literature. Int J Surg Pathol . 2011;19(2):212–216. doi:10.1177/1066896908320833 Morey AF, Meng MV, McAninch JW. Skin graft reconstruction of chronic genital lymphedema. Urology . 1997;50(3):423–426. doi:10.1016/S0090-4295(97)00259-8 Ndoye, Sylla, Ba, Guèye, Diagne. Management of penile and scrotal elephantiasis. BJU Int . 1999;84(3):362–364. doi:10.1046/j.1464-410x.1999.00223.x Aulia I, Yessica EC. Surgical management of male genital lymphedema: A systematic review. Arch Plast Surg . 2020;47(01):3–8. doi:10.5999/aps.2019.01123 Yamamoto T, Daniel BW, Rodriguez JR, et al. Radical reduction and reconstruction for male genital elephantiasis: Superficial circumflex iliac artery perforator (SCIP) lymphatic flap transfer after elephantiasis tissue resection. J Plast Reconstr Aesthet Surg . 2022;75(2):870–880. doi:10.1016/j.bjps.2021.08.011 Bulkley GJ. Scrotal and Penile Lymphedema. J Urol . 1962;87(3):422–429. doi:10.1016/S0022-5347(17)64975-9 Wisenbaugh E, Moskowitz D, Gelman J. Reconstruction of Massive Localized Lymphedema of the Scrotum: Results, Complications, and Quality of Life Improvements. Urology . 2018;112:176–180. doi:10.1016/j.urology.2016.09.063 Tekin HG, Andersen K, Bakholdt V, Sørensen JA. Resection and reconstruction of idiopathic scrotal elephantiasis: surgical approach and quality of life assessment. BMJ Case Rep . 2021;14(10):e244018. doi:10.1136/bcr-2021-244018 Haq AU, Afzal MO, Tarar MN. Giant Penoscrotal Lymphedema: Planning And Surgical Technique To Treat A Rare Debilitating Disease. J Ayub Med Coll Abbottabad JAMC . 2023;35(2):210–215. doi:10.55519/JAMC-02-10310 Welling H, Tsigka E, Krogh J, Schmidt VJ, Munksdorf M. Case report: surgical management of massive scrotal lymphedema in a bariatric patient. J Surg Case Rep . 2022;2022(4):rjac100. doi:10.1093/jscr/rjac100 Salako AA, Olabanji JK, Oladele AO, Alabi GH, Adejare IE, David RA. Surgical Reconstruction of Giant Penoscrotal Lymphedema in Sub-Saharan Africa. Urology . 2018;112:181–185. doi:10.1016/j.urology.2016.09.064 McGregor IA, Jackson IT. The Groin Flap. Br J Plast Surg . 1972;25(1):3–16. doi:10.1016/s0007-1226(72)80003-1 Abdelfattah U, Elbanoby T, Hamza F, et al. Treatment of Advanced Male Genital Lymphedema With a Complete Functional Lymphatic System Pedicled Transfer. Urology . 2023;175:190–195. doi:10.1016/j.urology.2023.02.006 Ciudad P, Escandón JM, Escandón L, Mayer HF, Manrique OJ. Surgical Management of Genital Lymphedema Using the Combined Charles’ Procedure and Lymphatic Superficial Circumflex Iliac Artery Perforator Flap Transfer (CHASCIP). Microsurgery . 2025;45(5):e70075. doi:10.1002/micr.70075 Additional Declarations No competing interests reported. Cite Share Download PDF Status: Under Revision Version 1 posted Editorial decision: Revision requested 25 Feb, 2026 Reviews received at journal 12 Feb, 2026 Reviewers agreed at journal 05 Feb, 2026 Reviews received at journal 03 Feb, 2026 Reviewers agreed at journal 03 Feb, 2026 Reviewers agreed at journal 03 Feb, 2026 Reviewers invited by journal 03 Feb, 2026 Editor assigned by journal 03 Feb, 2026 Submission checks completed at journal 31 Jan, 2026 First submitted to journal 31 Jan, 2026 You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. Our growing team is made up of researchers and industry professionals working together to solve the most critical problems facing scientific publishing. Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-8652655","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":585340568,"identity":"745adf31-9002-4631-9018-2f6218a40508","order_by":0,"name":"Alexander H. 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Patel","email":"","orcid":"","institution":"LKSOM Temple University Hospital/Fox Chase Cancer Center","correspondingAuthor":false,"prefix":"","firstName":"Sameer","middleName":"A.","lastName":"Patel","suffix":""},{"id":585340582,"identity":"5611c29f-9017-469e-8707-0bf14de09409","order_by":8,"name":"Andres Romero","email":"","orcid":"","institution":"Caja Costarricense de Seguro Social","correspondingAuthor":false,"prefix":"","firstName":"Andres","middleName":"","lastName":"Romero","suffix":""}],"badges":[],"createdAt":"2026-01-20 20:03:25","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-8652655/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-8652655/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":102182208,"identity":"a540f977-86d4-47ab-b1a0-281e389a7ef4","added_by":"auto","created_at":"2026-02-09 07:28:15","extension":"jpeg","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":191176,"visible":true,"origin":"","legend":"\u003cp\u003ePreoperative view with the hips flexed and abducted demonstrating \u0026nbsp;massive scrotal lymphedema with marked ulceration.\u003c/p\u003e","description":"","filename":"floatimage1.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-8652655/v1/bba8e54c06236bc63c9fa4db.jpeg"},{"id":102182210,"identity":"96012f03-23ef-4ef7-9c22-ae20400e2e80","added_by":"auto","created_at":"2026-02-09 07:28:15","extension":"jpeg","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":161037,"visible":true,"origin":"","legend":"\u003cp\u003eAnother intraoperative view demonstrating the extent and displacement \u0026nbsp;of lymphedematous mass during surgical preparation.\u003c/p\u003e","description":"","filename":"floatimage2.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-8652655/v1/abbd0219c8e3a5d5fe8b171a.jpeg"},{"id":102296687,"identity":"e7093565-2d98-47a2-bc08-efbd625b84c3","added_by":"auto","created_at":"2026-02-10 10:20:43","extension":"png","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":137871,"visible":true,"origin":"","legend":"\u003cp\u003ePreoperative coronal CT image, demonstrating massive scrotal \u0026nbsp;enlargement characterized by diffuse soft tissue thickening, subcutaneous edema, \u0026nbsp;and pronounced inferior extension of the lymphedematous mass.\u003c/p\u003e","description":"","filename":"floatimage3.png","url":"https://assets-eu.researchsquare.com/files/rs-8652655/v1/7625ccab8afb91a61b88fbe6.png"},{"id":102182212,"identity":"495d695e-00a7-425a-a4c5-00ec9eef9898","added_by":"auto","created_at":"2026-02-09 07:28:16","extension":"jpeg","order_by":4,"title":"Figure 4","display":"","copyAsset":false,"role":"figure","size":169267,"visible":true,"origin":"","legend":"\u003cp\u003eIntraoperative identification of the intact testes and spermatic cords\u003c/p\u003e","description":"","filename":"floatimage4.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-8652655/v1/d1b8b20c81a32059ad0e6865.jpeg"},{"id":102297143,"identity":"69c803be-8bb5-4bef-968b-6f6de466e0cb","added_by":"auto","created_at":"2026-02-10 10:26:04","extension":"jpeg","order_by":5,"title":"Figure 5","display":"","copyAsset":false,"role":"figure","size":154349,"visible":true,"origin":"","legend":"\u003cp\u003eImmediate postoperative view following wide excision and scrotal \u0026nbsp;reconstruction with the SCIP flap.\u003c/p\u003e","description":"","filename":"floatimage5.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-8652655/v1/79a399a39ccc70341a014168.jpeg"},{"id":102182207,"identity":"075352c3-5df5-4d14-8eab-7ccf4b4ce02f","added_by":"auto","created_at":"2026-02-09 07:28:15","extension":"png","order_by":6,"title":"Figure 6","display":"","copyAsset":false,"role":"figure","size":103652,"visible":true,"origin":"","legend":"\u003cp\u003eDiagram outlining the screening, exclusion, and final selection process for studies included in the review.\u003c/p\u003e","description":"","filename":"floatimage6.png","url":"https://assets-eu.researchsquare.com/files/rs-8652655/v1/112ab437b21dc8953f7a522e.png"},{"id":102299367,"identity":"74a0b297-2751-4529-842b-c399d435ae6f","added_by":"auto","created_at":"2026-02-10 11:05:01","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":1425341,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-8652655/v1/1024ee8e-9d4d-4b34-8b7a-b0391fe79971.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Surgical Management of Massive Scrotal Lymphedema: A Case Report and Review of the Literature","fulltext":[{"header":"Introduction","content":"\u003cp\u003eLymphedema is a chronic, progressive disorder characterized by impaired lymphatic drainage and accumulation of protein-rich interstitial fluid within soft tissues, leading to inflammation, fibrosis, and progressive hypertrophy of the affected region.\u003csup\u003e1\u003c/sup\u003e Recurrent lymphatic obstruction triggers chronic inflammation, fibroblast proliferation, and adipose deposition, resulting in irreversible tissue thickening and deformity.\u003csup\u003e2\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eAlthough most commonly involving the extremities, genital lymphedema can cause profound functional disability and psychosocial distress.\u003csup\u003e3\u003c/sup\u003e Lymphedema involving the genitalia can be classified as primary, due to congenital lymphatic dysplasia, or secondary, arising from infection, malignancy, trauma, obesity, or previous surgical or radiation injury.\u003csup\u003e4,5\u003c/sup\u003e In endemic regions such as sub-Saharan Africa, Southeast Asia, and parts of South America, filarial infection remains the leading cause of genital lymphedema.\u003csup\u003e2,3\u003c/sup\u003e In nonendemic areas, chronic inflammatory, neoplastic, or iatrogenic mechanisms predominate.\u003csup\u003e5\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eProgressive scrotal involvement may result in massive enlargement, recurrent infection, and significant impairment of mobility, hygiene, urination, and sexual activity. In severe cases, the mass effect can become debilitating, causing restriction in daily activities and psychosocial distress.\u003csup\u003e1\u003c/sup\u003e The underlying tissue changes include fibrosis, dermal thickening, and lymphangiectasia, which render conservative measures such as compression, massage, and antibiotics largely ineffective once fibrosis becomes established.\u003csup\u003e5,6\u003c/sup\u003e Surgical excision of diseased tissue with preservation of viable structures remains the definitive treatment for chronic or massive lymphedema.\u003csup\u003e7,8\u003c/sup\u003e The primary goals of surgery are to restore penile and scrotal anatomy, reduce infection risk, and re-establish urinary and sexual function.\u003csup\u003e4\u003c/sup\u003e Successful outcomes depend on accurate preoperative mapping, complete removal of fibrotic tissue, and tension-free reconstruction that provides thin, pliable coverage while minimizing donor site morbidity.\u003csup\u003e1,5\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eMultiple surgical techniques have been described for genital lymphedema, and the choice of approach depends on disease severity, tissue viability, and reconstructive goals. Local excision with primary closure may suffice for limited disease, whereas advanced cases often require split-thickness skin grafting or flap coverage to achieve durable, pliable reconstruction.\u003csup\u003e3,9\u003c/sup\u003e Flaps such as the gracilis, anterolateral thigh (ALT), and superficial circumflex iliac artery (SCIA) or superficial circumflex iliac artery perforator (SCIP) flaps provide well-vascularized tissue suitable for resurfacing and functional restoration.\u003csup\u003e10\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eThis case report describes the management of massive scrotal lymphedema complicated by a buried penis and severe reduction in urinary and sexual function. The magnitude of disease required comprehensive diagnostic evaluation to exclude infectious, neoplastic, and vascular causes and to delineate the extent of fibrotic and viable tissue. Precise preoperative mapping was critical to identify preserved penile and testicular structures, guide resection margins, and anticipate the reconstructive requirements.\u003c/p\u003e"},{"header":"Case","content":"\u003cp\u003eA 52-year-old man from a tropical region presented with a 10-year history of progressive scrotal enlargement. He reported increasing difficulty with activities of daily living. Walking and sitting were particularly challenging, as the scrotum extended to his lower legs and rested on the floor when he stood upright (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003e). The mass had become ulcerated, placing him at risk for systemic infection. The scrotum weighed approximately 30 kg and completely buried the penis within the hypertrophic folds, resulting in multiple urinary tract infections over the years (Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003e).\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eChronic inflammation and recurrent infection led to urethral strictures, ultimately preventing complete evacuation of urine. Despite severe physical and psychological distress, the patient had not sought medical attention earlier due to financial limitations and poor access to definitive surgical treatment.\u003c/p\u003e \u003cp\u003eThe patient had no significant past medical history. On examination, the scrotum was grossly enlarged, extending to the level of the calves, with thickened, indurated skin consistent with elephantiasis. Physical examination also revealed a buried penis with loss of sexual function. The presence of multiple urethral strictures, a collapsed urethral lumen, and massive scrotal bulk precluded urinary evacuation. Filariasis was excluded early in the diagnostic process through negative peripheral smear results and the absence of systemic manifestations. Multiple punch biopsies revealed nonspecific chronic inflammation and dermal fibrosis without evidence of malignancy or infection. Ultrasound and CT imaging ruled out underlying masses. Pelvic CT demonstrated markedly dilated lymphatic channels and diffuse thickening of the scrotal tissue, confirming the diagnosis of idiopathic non-filarial scrotal lymphedema (Fig.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e3\u003c/span\u003e).\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eCT angiography showed no significant arterial or venous compromise. Given the profound anatomic distortion and total loss of function, surgical intervention was deemed necessary.\u003c/p\u003e \u003cp\u003eA multidisciplinary team proceeded with a two-stage surgical approach. The first stage focused on debulking and excision of redundant scrotal tissue to expose the penis and testes, followed by definitive reconstruction. A horizontal incision was made in the groin area, with the anatomical outline premarked to map the skin for future defect coverage. The size and extent of the incision were determined by the anticipated defect. Careful undermining of the abdominal wall was performed to preserve perforating arteries within the abdominal tissue. Skin dissection was carried out to facilitate wound approximation following excision of the excessive scrotal tissue. Dissection continued distally to the pubis, with the goal of identifying and assessing the phallus.\u003c/p\u003e \u003cp\u003eAt this stage, the urology team assumed operative management for tissue salvage and revision of urethral strictures and testicular anatomy. Urethroscopy was performed for stricture release, followed by placement of a permanent Foley catheter. The testes and spermatic cords were inspected and found to be anatomically and functionally preserved (Fig.\u0026nbsp;\u003cspan refid=\"Fig4\" class=\"InternalRef\"\u003e4\u003c/span\u003e).\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eThe second and final stage was performed by the plastic surgery team. A meticulous perineal dissection was carried out, with emphasis on identifying the anterior and posterior tissue boundaries to guide bilateral flap design. A superficial circumflex iliac artery perforator (SCIP) flap was selected to cover the genitals and protect the exposed spermatic cords. The anterior superior iliac spine and pubic tubercle served as key landmarks during flap harvest. After incising the deep fascia, subfascial dissection was performed to identify the superficial circumflex iliac artery, which was traced to its origin from the femoral artery, allowing mobilization of the pedicle for posterior rotation of the flap. The deeper perineal layers were closed using absorbable monofilament sutures, while the superficial dermal layer was closed with Vicryl. Multiple drains were placed for lymphatic drainage and fluid management.\u003c/p\u003e \u003cp\u003eTo optimize functional outcomes, a Z-plasty was performed at the penile base following release, and full-thickness skin grafts harvested from the inner thigh were used for shaft reconstruction (Fig.\u0026nbsp;\u003cspan refid=\"Fig5\" class=\"InternalRef\"\u003e5\u003c/span\u003e).\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003ePostoperative recovery was uneventful. No wound dehiscence or revision was required. The patient was able to void normally after reconstruction. At a 3-month follow-up, the patient reported marked improvement in mobility, hygiene, and quality of life.\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eScrotal lymphedema is a rare and debilitating condition characterized by chronic lymphatic obstruction leading to progressive enlargement and fibrosis of the scrotal tissues.\u003csup\u003e1\u003c/sup\u003e Although most cases in tropical regions are caused by filarial infection, this patient\u0026rsquo;s presentation was non-filarial, supported by negative parasitological and serologic testing.\u003csup\u003e2\u003c/sup\u003e Non-filarial genital lymphedema may arise secondary to neoplastic, inflammatory, or idiopathic causes.\u003csup\u003e5\u003c/sup\u003e The chronicity of this case and absence of secondary pathology support a diagnosis of idiopathic non-filarial scrotal lymphedema.\u003c/p\u003e \u003cp\u003eThe earliest detailed description of surgical management dates to 1820, when Professor Jacques-Mathieu Delpech of Montpellier performed radical excision of a 60-pound scrotal mass in Jean Baptiste Authier, a cavalryman disabled by syphilitic lymphatic obstruction.\u003csup\u003e11\u003c/sup\u003e The operation was carried out without anesthesia before an audience of 300, establishing the enduring principles of complete resection and local flap reconstruction later depicted in Delpech\u0026rsquo;s operative drawings.\u003csup\u003e11\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eSubsequent surgeons, including Matas (1913), Young (1926), and DeSavitsch (1941) refined these methods while maintaining the same operative philosophy. Together, these historical precedents illustrate that despite technical advances over two centuries, the fundamental principle has remained unchanged: radical excision of diseased tissue with tension-free, well-vascularized coverage remains the cornerstone of treatment for genital elephantiasis.\u003c/p\u003e \u003cp\u003eTo contextualize our case within the broader literature, a comprehensive review was performed. A total of 350 articles were identified, of which 60 met inclusion criteria for studies addressing surgical management of scrotal lymphedema. These included 31 case reports, 25 case series, and 4 literature reviews published between 1977 and 2025. The screening and selection process is summarized in Fig.\u0026nbsp;\u003cspan refid=\"Fig6\" class=\"InternalRef\"\u003e6\u003c/span\u003e.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eOur case presentation of a 32 kg resection ranks among the largest documented in the last decades, with reported resection weights across the literature ranging from 0.9 to 61 kg. Representative modern series are summarized in Table\u0026nbsp;\u003cspan refid=\"Tab1\" class=\"InternalRef\"\u003e1\u003c/span\u003e. Wisenbaugh et al. described the heaviest excision to date (61 kg) managed by wide en bloc resection and primary closure using preserved lateral and posterior scrotal flaps.\u003csup\u003e12,13\u003c/sup\u003e Tekin et al. describes surgical management of a 42 kg idiopathic case through subtotal scrotectomy and posterior flap advancement, avoiding grafting entirely. Haq et al.\u003csup\u003e14\u003c/sup\u003e reported a 33 kg resection managed with vacuum-assisted closure and delayed split-thickness grafting, while Welling et al. describes staged debulking and resurfacing in a morbidly obese patient with a BMI of 77, achieving full functional recovery.\u003csup\u003e15\u003c/sup\u003e Salako et al. used a modified Charles procedure for a 22.4 kg case, employing preserved preputial and scrotal-neck skin for primary closure.\u003csup\u003e16\u003c/sup\u003e\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab1\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 1\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003eReported cases of massive scrotal or penoscrotal lymphedema requiring large-volume excision, including operative technique and resection weight. Note * was the reported average, not the maximum singular weight. Across these large resections, outcomes consistently favored complete excision with primary closure using viable local tissue over graft-only repairs, with lower complication rates and superior functional results. When native skin was insufficient, split-thickness skin grafts (STSG) provided temporary coverage but were prone to contracture and breakdown. Consequently, regional and pedicled flaps, most commonly based on the superficial circumflex iliac artery (SCIA), anterolateral thigh (ALT), and gracilis flaps, became the preferred reconstructive options, offering thin, vascularized coverage and reduced recurrence as summarized in Table\u0026nbsp;\u003cspan refid=\"Tab2\" class=\"InternalRef\"\u003e2\u003c/span\u003e.\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"3\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAuthor (Year)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003eResection Technique\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eWeight of Resection (kg)\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eWisenbaugh et al., 2018\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eEn bloc excision of massive, localized lymphedema with preservation of viable lateral and posterior scrotal flaps for primary closure, avoiding grafts when possible\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e61\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTekin et al., 2021\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eSubtotal scrotectomy performed emergently for hemorrhagic idiopathic scrotal elephantiasis; excision extended through lateral folds to posterior wall\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e42\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eThejeswi et al., 2012\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eNear-total excision of scrotal skin; circumferential penile dissection; debulking using cautery and Ligasure; bilateral orchiectomy due to atrophy\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e32\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eWelling et al., 2022\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eTwo-stage excision and reconstruction in a morbidly obese (BMI 77) patient; initial wide debulking followed by delayed penile resurfacing\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e26\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eSalako et al., 2018\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eModified Charles procedure (i.e., complete excision of all diseased penoscrotal tissue to Buck\u0026rsquo;s fascia with immediate reconstruction using preserved healthy preputial and scrotal-neck skin for primary closure over a drain)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e22.4*\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eHaq et al., 2023\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eStaged excision of giant penoscrotal lymphedema using vacuum-assisted closure (VAC) to control edema and prepare wound bed for delayed grafting\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e20\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab2\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 2\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003eTable highlighting reconstruction techniques of largest reported mass resections.\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"4\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c4\" colnum=\"4\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e \u003cp\u003eAuthor (Year)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003ePenile Reconstruction\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eScrotal Reconstruction\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003eClosure\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eWisenbaugh et al., 2018\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eNative penile skin preserved; grafting deferred to minimize breakdown risk\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003eLateral and posterior flaps advanced medially to reconstitute scrotum\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003ePrimary closure with drains\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eTekin et al., 2021\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eBuried penis reconstructed using residual preputial skin; STSG considered but not required\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003ePosterior scrotal wall advanced and folded medially to create neoscrotum\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003ePrimary closure without drains\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eThejeswi et al., 2012\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eBilateral scrotal skin flaps advanced with Y-shaped midline closure\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003eNative penile skin preserved; no grafts or distant flaps used\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003ePrimary closure using preserved Y-shaped scrotal flaps\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eWelling et al., 2022\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003ePenile shaft left degloved initially, resurfaced secondarily with STSG\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003eLateral scrotal flaps preserved and advanced medially to form neoscrotum\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003eTwo-stage closure\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eSalako et al., 2018\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eUninvolved preputial skin rolled back along shaft for penile resurfacing\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003eNeoscrotum fashioned from stretched healthy neck skin; tunica vaginalis everted around testes\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003ePrimary closure with drain; scrotal support post-op\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eHaq et al., 2023\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003eSplit-thickness skin graft (STSG) applied after edema resolution\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003eLocal medial and posterior skin flaps preserved for coverage and contouring\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003eDelayed closure following VAC therapy\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cp\u003eThe SCIP flap used in our case represents the modern evolution of the classical McGregor groin flap. McGregor and Jackson originally described a pedicled groin flap based on the SCIA and SIEA systems for upper extremity coverage, and their seminal paper highlighted its reliability but also noted its bulk and limited contour precision.\u003csup\u003e17\u003c/sup\u003e Subsequent refinements of the SCIA system, including the development of perforator-based SCIA and SCIP designs, have produced thin, pliable, and well-vascularized skin with minimal donor-site morbidity. These advances make contemporary SCIA-based flaps particularly well suited for genital and perineal resurfacing.\u003c/p\u003e \u003cp\u003eWhile the SCIA and SCIP flaps share a vascular origin, they serve different reconstructive goals. The SCIA flap, derived from the classical groin or McGregor flap, provides a reliable pedicled option with thicker and more robust fasciocutaneous tissue suitable for larger perineal, suprapubic, or inguinal defects and for cases where microsurgery is not feasible or the wound bed is compromised. In contrast, the SCIP flap offers thin, pliable tissue with minimal donor-site morbidity, making it well suited for genital, perineal, and distal extremity resurfacing where contour and precision are critical. In our case, the extent and location of the defect necessitated a thin flap transfer, for which the SCIP flap provided superior functional and aesthetic outcomes.\u003c/p\u003e \u003cp\u003eAcross the modern literature, outcomes following SCIA-based reconstruction are consistently favorable, with most patients achieving restored urinary function, improved hygiene, and enhanced mobility within three to six months, and only minor wound dehiscence or seroma formation reported. As perforator and lymphatic-restorative techniques continue to evolve, the SCIA and SCIP flaps remain among the most reliable and reproducible regional options for definitive reconstruction after massive genital lymphedema.\u003c/p\u003e \u003cp\u003eAlthough disruption of lymphatic drainage has long been recognized, with early attempts including permanent suturing of the scrotum to the thigh to facilitate alternative drainage pathways, reconstructive options at the time lacked the technical means to reliably address this problem.\u003csup\u003e11\u003c/sup\u003e With the advancement of microsurgical techniques, newer options have expanded the reconstructive armamentarium. Perforator flaps as well as lymphatic-restorative flaps vascularized lymph node transfers, provide more physiologic coverage while potentially reestablishing lymphatic drainage.\u003csup\u003e10,18,19\u003c/sup\u003e These innovations reflect a paradigm shift from purely excisional to functionally restorative reconstruction, particularly in centers with microsurgical capability.\u003c/p\u003e \u003cp\u003eOur case resonates with the literature in that wide excision combined with vascularized regional tissue coverage remains the cornerstone of management. Microsurgical and lymphatic-restorative techniques represent promising advances; however, access to these treatments remains limited, particularly in low- and middle-income countries (LMICs), and long-term outcome data are sparse. The pedicled SCIA flap and its descendant the SCIP remain the most versatile, reproducible, and globally applicable options for definitive reconstruction following massive scrotal lymphedema.\u003c/p\u003e"},{"header":"Declarations","content":" \u003cp\u003e \u003cstrong\u003eEthics approval and consent to participate:\u003c/strong\u003e \u003cp\u003eAll ethics protocol for institutions were observed; patient\u0026rsquo;s consent for images to be used for publication is documented.\u003c/p\u003e \u003c/p\u003e \u003cp\u003e \u003cstrong\u003eClinical trial number\u003c/strong\u003e \u003cp\u003eNot applicable\u003c/p\u003e \u003c/p\u003e \u003cp\u003e \u003cstrong\u003eConsent for publication:\u003c/strong\u003e \u003cp\u003ePhotographs were taken with consent; written informed consent for publication of their clinical details and/or clinical images was obtained from the patient. A copy of the consent form is available for review by the Editor of this journal.\u003c/p\u003e \u003c/p\u003e\u003cp\u003e \u003ch2\u003eCompeting interests:\u003c/h2\u003e \u003cp\u003eThe author declares no competing interests.\u003c/p\u003e \u003c/p\u003e\u003cp\u003e \u003ch2\u003eAcknowledgements\u003c/h2\u003e \u003cp\u003eNot applicable.\u003c/p\u003e \u003c/p\u003e\u003ch2\u003eFunding:\u003c/h2\u003e \u003cp\u003eNone.\u003c/p\u003e\u003ch2\u003eAuthor Contribution\u003c/h2\u003e\u003cp\u003eAlexander H. Chang, Natalia Mejia, Aryan Gupta, Jing Qin Tay, Ambroise Gilles, Sthefano Araya, Adam C. Walchak, Sameer A. Patel, Andres Romero have all contributed equally to the manuscript.\u003c/p\u003e\u003ch2\u003eData Availability\u003c/h2\u003e\u003cp\u003eNot applicable.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003eMcDougal WS. Lymphedema of the External Genitalia. \u003cem\u003eJ Urol\u003c/em\u003e. 2003;170(3):711–716. doi:10.1097/01.ju.0000067625.45000.9e\u003c/li\u003e\n\u003cli\u003eDandapat MC, Mohapatro SK, Patro SK. Elephantiasis of the Penis and Scrotum. A Review of 350 Cases. \u003cem\u003eAm J Surg\u003c/em\u003e. 1985;149(5):686–690. doi:10.1016/s0002-9610(85)80156-2\u003c/li\u003e\n\u003cli\u003eKaciulyte J, Garutti L, Spadoni D, et al. Genital Lymphedema and How to Deal with It: Pearls and Pitfalls from over 38 Years of Experience with Unusual Lymphatic System Impairment. \u003cem\u003eMedicina (Mex)\u003c/em\u003e. 2021;57(11):1175. doi:10.3390/medicina57111175\u003c/li\u003e\n\u003cli\u003eHolman CM, Arnold PG, Jurkiewicz MJ, Walton KN. Reconstruction of Male External Genitalia with Elephantiasis. \u003cem\u003eUrology\u003c/em\u003e. 1977;10(6):576–578. doi:10.1016/0090-4295(77)90108-X\u003c/li\u003e\n\u003cli\u003eBrewer MB, Singh DP. Massive Localized Lymphedema: Review of an Emerging Problem and Report of a Complex Case in the Mons Pubis. \u003cem\u003eAnn Plast Surg\u003c/em\u003e. 2012;68(1):101–104. doi:10.1097/SAP.0b013e318211910e\u003c/li\u003e\n\u003cli\u003eBogusz AM, Hussey SM, Kapur P, Yan Peng, Tunc Gokaslan S. Massive Localized Lymphedema With Unusual Presentations: Report of 2 Cases and Review of the Literature. \u003cem\u003eInt J Surg Pathol\u003c/em\u003e. 2011;19(2):212–216. doi:10.1177/1066896908320833\u003c/li\u003e\n\u003cli\u003eMorey AF, Meng MV, McAninch JW. Skin graft reconstruction of chronic genital lymphedema. \u003cem\u003eUrology\u003c/em\u003e. 1997;50(3):423–426. doi:10.1016/S0090-4295(97)00259-8\u003c/li\u003e\n\u003cli\u003eNdoye, Sylla, Ba, Guèye, Diagne. Management of penile and scrotal elephantiasis. \u003cem\u003eBJU Int\u003c/em\u003e. 1999;84(3):362–364. doi:10.1046/j.1464-410x.1999.00223.x\u003c/li\u003e\n\u003cli\u003eAulia I, Yessica EC. Surgical management of male genital lymphedema: A systematic review. \u003cem\u003eArch Plast Surg\u003c/em\u003e. 2020;47(01):3–8. doi:10.5999/aps.2019.01123\u003c/li\u003e\n\u003cli\u003eYamamoto T, Daniel BW, Rodriguez JR, et al. Radical reduction and reconstruction for male genital elephantiasis: Superficial circumflex iliac artery perforator (SCIP) lymphatic flap transfer after elephantiasis tissue resection. \u003cem\u003eJ Plast Reconstr Aesthet Surg\u003c/em\u003e. 2022;75(2):870–880. doi:10.1016/j.bjps.2021.08.011\u003c/li\u003e\n\u003cli\u003eBulkley GJ. Scrotal and Penile Lymphedema. \u003cem\u003eJ Urol\u003c/em\u003e. 1962;87(3):422–429. doi:10.1016/S0022-5347(17)64975-9\u003c/li\u003e\n\u003cli\u003eWisenbaugh E, Moskowitz D, Gelman J. Reconstruction of Massive Localized Lymphedema of the Scrotum: Results, Complications, and Quality of Life Improvements. \u003cem\u003eUrology\u003c/em\u003e. 2018;112:176–180. doi:10.1016/j.urology.2016.09.063\u003c/li\u003e\n\u003cli\u003eTekin HG, Andersen K, Bakholdt V, Sørensen JA. Resection and reconstruction of idiopathic scrotal elephantiasis: surgical approach and quality of life assessment. \u003cem\u003eBMJ Case Rep\u003c/em\u003e. 2021;14(10):e244018. doi:10.1136/bcr-2021-244018\u003c/li\u003e\n\u003cli\u003eHaq AU, Afzal MO, Tarar MN. Giant Penoscrotal Lymphedema: Planning And Surgical Technique To Treat A Rare Debilitating Disease. \u003cem\u003eJ Ayub Med Coll Abbottabad JAMC\u003c/em\u003e. 2023;35(2):210–215. doi:10.55519/JAMC-02-10310\u003c/li\u003e\n\u003cli\u003eWelling H, Tsigka E, Krogh J, Schmidt VJ, Munksdorf M. Case report: surgical management of massive scrotal lymphedema in a bariatric patient. \u003cem\u003eJ Surg Case Rep\u003c/em\u003e. 2022;2022(4):rjac100. doi:10.1093/jscr/rjac100\u003c/li\u003e\n\u003cli\u003eSalako AA, Olabanji JK, Oladele AO, Alabi GH, Adejare IE, David RA. Surgical Reconstruction of Giant Penoscrotal Lymphedema in Sub-Saharan Africa. \u003cem\u003eUrology\u003c/em\u003e. 2018;112:181–185. doi:10.1016/j.urology.2016.09.064\u003c/li\u003e\n\u003cli\u003eMcGregor IA, Jackson IT. The Groin Flap. \u003cem\u003eBr J Plast Surg\u003c/em\u003e. 1972;25(1):3–16. doi:10.1016/s0007-1226(72)80003-1\u003c/li\u003e\n\u003cli\u003eAbdelfattah U, Elbanoby T, Hamza F, et al. Treatment of Advanced Male Genital Lymphedema With a Complete Functional Lymphatic System Pedicled Transfer. \u003cem\u003eUrology\u003c/em\u003e. 2023;175:190–195. doi:10.1016/j.urology.2023.02.006\u003c/li\u003e\n\u003cli\u003eCiudad P, Escandón JM, Escandón L, Mayer HF, Manrique OJ. Surgical Management of Genital Lymphedema Using the Combined Charles’ Procedure and Lymphatic Superficial Circumflex Iliac Artery Perforator Flap Transfer (CHASCIP). \u003cem\u003eMicrosurgery\u003c/em\u003e. 2025;45(5):e70075. doi:10.1002/micr.70075\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"bmc-plastic-and-reconstructive-surgery","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"","sideBox":"Learn more about [BMC Plastic and Reconstructive Surgery](https://link.springer.com/journal/44452)","snPcode":"44452","submissionUrl":"https://submission.springernature.com/new-submission/44452/3","title":"BMC Plastic and Reconstructive Surgery","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"stoa","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"","lastPublishedDoi":"10.21203/rs.3.rs-8652655/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-8652655/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003eScrotal lymphedema, or scrotal elephantiasis, is a rare progressive condition caused by impaired lymphatic drainage and accumulation of protein-rich interstitial fluid, resulting in fibrosis and massive genital enlargement. This case report presents a 52-year-old man with massive idiopathic scrotal lymphedema, or scrotal elephantiasis, characterized by a 10-year history of progressive scrotal enlargement, recurrent infections, and loss of urinary and sexual function with a completely buried penis Diagnostic evaluation excluded infectious, neoplastic, and vascular causes, confirming non-filarial scrotal lymphedema. Management consisted of staged radical excision of fibrotic tissue with preservation of the testes, spermatic cords, and phallus, followed by reconstruction using a pedicled superficial circumflex iliac artery perforator groin flap. Adjunctive Z-plasty at the penile base and full-thickness thigh skin grafts restored contour and mobility. Postoperative recovery was uneventful, with complete flap survival and restoration of urinary function, hygiene, mobility, and quality of life. This case demonstrates that radical excision with SCIP flap reconstruction provides a reliable and durable solution for massive idiopathic scrotal lymphedema, including cases requiring tissue resection exceeding 30 kg. 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