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A 61-year-old man with a history of atrioventricular block and perforated ulcer was referred for evaluation of enlarging growths on his testicle. These growths were first noted approximately 6 months ago and are itchy at times. The patient denies any bleeding or pain related to the growths. Physical examination reveals a 3-cm firm, nonulcerated, multilobular tumor with variegate coloration.
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Buschke-Löwenstein tumor (BLT) is a type of verrucous carcinoma first described by Buschke and Löwenstein in 1925.1 It is caused by the human papillomavirus (HPV), in particular by HPV serotypes 6 and 11, and is always preceded by condyloma acuminata.2
The carcinoma is rare, with an incidence among sexually active adults of approximately 0.1% per year.2 It can occur at any age after puberty but mainly is seen among adults aged 40 and 60 years.2 Although HPV infection affects both men and women, the incidence in men predominates.2 Immunosuppression, chronic inflammation, poor hygiene, and coinfection with HIV are likely to be risk factors for the development of BLT.2
BLT is a variant of squamous cell carcinoma (SCC) that most commonly appears on the glans penis, foreskin, or perianal regions.1 It typically begins as a small keratotic papule that over several years morphs into a cauliflower-like mass with cutaneous fistulas and a cobblestone surface. 1 The size of the growth may exceed 10 cm.3
Histopathologically, BLT exhibits a densely keratinized and well-differentiated epithelium. Structures similar to those seen in condyloma acuminata are noted such as vacuolization of the cells in the superficial layer of the epidermis, marked degree of acanthosis and parakeratosis, and infrequent mitotic figures.3 BLT is differentiated from other growths by its thicker stratum corneum, marked papillary proliferations, and its tendency to invade deeply, displacing the underlying tissues.4 Differentiation between BLT and malignant SCC is based on the absence of invasion, intact basement membrane, infrequent mitotic figures, and no development of metastases. Since the description of this entity, there have been numerous reports of SCC coexisting with the BLT.4
Mohs surgery is a preferred treatment modality as it allows for tissue sparing while enabling the surgeon to ensure tumor-free margins and recognize foci of SCC.5 If necessary, surgical defects can later be surgically reconstructed. 6 BLT has a high rate of recurrence.5 In most cases, BLT recurrences are managed by radical surgery or carbon dioxide/argon laser resection.7
Topical, intralesional, and oral chemotherapy have been used as adjuvant therapy and for treating tumor recurrences after surgery.8,9 Chemotherapy alone, however, has not shown promising results.10 Radiation therapy, in spite of its neoadjuvant application in a limited number of BLT cases, has largely been avoided, mostly because of the fear of causing further malignant transformations.11,12
Dorsaf Elinkichari, MD, Faten Rabhi, MD, Kahena Jaber, MD, and Mohamed Raouf Dhaoui, MD, are from the Department of Dermatology, Military Hospital of Tunis, Tunisia. Stephen Schleicher, MD, is director of the DermDox Center for Dermatology in Pennsylvania, as well as an associate professor of medicine at Commonwealth Medical College and a clinical instructor of dermatology at Arcadia University and Kings College.
References
1. Irshad U, Puckett Y. Giant condylomata acuminata of Buschke and Löwenstein. In: StatPearls. Treasure Island: StatPearls Publishing; 2021.
2. Kadouri Y, Nouini Y. Buschke-Löwenstein´ tumor. Pan Afr Med J. 2020;36:359. doi:10.11604/pamj.2020.36.359.13292
3. Balázs M. Buschke-Loewenstein tumour. A histologic and ultrastructural study of six cases. Virchows Arch A Pathol Anat Histopathol. 1986;410(2):83-92. doi:10.1007/BF00713509
4. Chu QD, Vezeridis MP, Libbey NP, Wanebo HJ. Giant condyloma acuminatum (Buschke-Löwenstein tumor) of the anorectal and perianal regions. Analysis of 42 cases. Dis Colon Rectum. 1994;37(9):950-957. doi:10.1007/BF02052606
5. Renzi A, Giordano P, Renzi G, Landolfi V, Del Genio A, Weiss EG. Buschke-Löwenstein tumor successful treatment by surgical excision alone: a case report. Surg Innov. 2006;13(1):69-72. doi:10.1177/155335060601300111
6. Balik E, Eren T, Bugra D. A surgical approach to anogenital Buschke Loewenstein tumours (giant condyloma acuminata). Acta Chir Belg. 2009;109(5):612-616. doi:10.1080/00015458.2009.11680497
7. Perniola G, d’Itri F, Di Donato V, Achilli C, Lo Prete E, Panici PB. Recurrent Buschke-Löwenstein tumor treated using CO (2) laser vaporization. J Minim Invasive Gynecol. 2010;17(5):662-664. doi:10.1016/j.jmig.2010.05.007
8. Dinleyici M, Saracoglu N, Eren M, et al. Giant condyloma acuminate due to human papillomavirus type 16 in an infant successfully treated with topical imiquimod therapy. Dermatol Reports. 2015;7(3):6134. doi:10.4081/dr.2015.6134
9. Toro JR, Sanchez S, Turiansky G, Blauvelt A. Topical cidofovir for the treatment of dermatologic conditions: verruca, condyloma, intraepithelial neoplasia, herpes simplex and its potential use in smallpox. Dermatol Clin. 2003;21(2):301-309. doi:10.1016/s0733-8635(02)00116-x
10. Ilkay AK, Chodak GW, Vogelzang NJ, Gerber GS. Buschke-Löwenstein tumor: therapeutic options including systemic chemotherapy. Urology. 1993;42(5):599-602. doi:10.1016/0090-4295(93)90288-l
11. Butler TW, Gefter J, Kleto D, Shuck EH, Ruffner BW. Squamous-cell carcinoma of the anus in condyloma acuminatum. Successful treatment with preoperative chemotherapy and radiation. Dis Colon Rectum. 1987;30(4):293-295. doi:10.1007/BF02556178
12. Sobrado CW, Mester M, Nadalin W, Nahas SC, Bocchini SF, Habr-Gama A. Radiation-induced total regression of a highly recurrent giant perianal condyloma: report of case. Dis Colon Rectum. 2000;43(2):257-260. doi:10.1007/BF02236991
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