107 Oncology, Gastroenterology and Hepatology Reports| Jul-Dec 2015 | Vol 4 | Issue 2

Priyanka Aggarwal, Gaurav Aggarwal1

Department of Dermatology, Dr. D. Y. Patil Medical College,

Pune, Maharashtra, 1Department of Surgical Oncology, Tata Medical

Center, Newtown, Kolkata, West Bengal, India

Address for the Correspondence:Dr. Gaurav Aggarwal,

Department of Surgical Oncology, Tata Medical Center,

Newtown, Kolkata ‑ 700 156, West Bengal, India.

E‑mail: drgaurav1981@rediffmail.com

Buschke–Lowenstein tumor of the inguinal region: A rare entity in a rarer location

Buschke–Lowenstein tumor (BLT), also known as giant condyloma accuminata, first described in 1896 by Buschke, is a rare, slow‑growing, locally invasive, mass lesion commonly affecting the anogenital region in immunocompromised males. This florid type of verrucous carcinoma is known for its high rate of recurrence as well as malignant conversion to squamous cell carcinoma. BLTs have been commonly reported to involve the penis, scrotum, bladder, vulva and perianal or anorectal regions. Our index case remains distinguished on the account of the rare anatomical location, causing a diagnostic dilemma, and once diagnosed managed adequately via surgery alone.

Key words: Buschke–Lowenstein tumor, giant condyloma acuminata, inguinal region

INTRODUCTION

Buschke–Lowenstein tumor (BLT), also known as giant condyloma accuminata, first described in 1896 by Buschke,[1] is a rare, slow‑growing, locally invasive, mass lesion commonly affecting the anogenital region in immunocompromised male patients.[2] This florid type of verrucous carcinoma is known for its high rate of recurrence as well as malignant conversion to squamous cell carcinoma.[3] BLT has been most commonly reported to involve the penis, scrotum, bladder, vulva and perianal or anorectal regions.[3] Our case remains distinguished on the account of the rare anatomical location, causing a diagnostic dilemma, and once diagnosed managed adequately by surgery alone.

CASE REPORT

A 48‑year‑old male of a lower socioeconomic status presented to the outpatient department with a giant warty lesion in his right inguinal area, which had slowly grown to its current size over a period of 18 years. The lesion was a solitary exophytic cauliflower like mass with an irregular surface, of size 12 cm × 10 cm in dimensions [Figure 1]. A foul odour with putative discharge was eminent which hampered his social activities. Superficial inguinal lymph nodes were nonpalpable, and there were no signs of satellitism or local tumor infiltration, or similar lesions elsewhere in the body. History and general physical examination were unremarkable, and seronegativity was confirmed by ELISA.

An incisional biopsy was taken, and results were consistent with condyloma accuminata.

Wide local excision with 2 cm margins with intraoperative frozen section confirmation of margin negativity and primary wound closure was performed [Figure 2] and the specimen was sent for histopathology.

The final histopathology also confirmed with the diagnosis of BLT [Figure 3]. The 3 monthly follow‑ups till 1‑year postoperatively were uneventful with no evidence of recurrence of the lesion.

DISCUSSION

Buschke–Lowenstein tumor in conjunction with Ackerman tumor, oral papillomatosis and papillomatosis cutis calcinoides encompasses a group of premalignant disorders that evolve over a long span of time but have a considerable potential for malignant transformation to squamous cell carcinomas (40–60%).[3,4]

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Aggarwal and Aggarwal: Buschke-Lowenstein tumour of the inguinal region

108Oncology, Gastroenterology and Hepatology Reports| Jul-Dec 2015 | Vol 4 | Issue 2

Buschke–Lowenstein tumors can be differentiated from the usual condylomas as being larger, nonmetastasizing and usually unresponsive to conservative management.[5] They are seen to affect males more than females with an average age of presentation at 43 years.[6] Various risk factors have been implicated such as immunosuppression, chronic irritation from perianal fistulas and

ulcerative colitis, poor personal hygiene as well as an association with human papilloma virus 6 and 11.[5]

Buschke–Lowenstein tumors may be seen as peri‑prepucial warty lesions as well as in the anal canal giving rise to fistulae emanating purulent discharging material.[7] This subsequently leads to local gigantism of the genitalia with condylomatous lesions.[7] Microscopically, BLTs form a mass of well‑differentiated squamous cells with no cellular anaplasia, surrounded by an acute or chronic inflammatory infiltrate.[7]

Radical surgery remains the gold standard in the management of a giant condyloma acuminata[8] taking into consideration its size, site as well as the possibility of defect closure thereafter. Various chemotherapeutic agents such as interferon‑2 alpha, 5 fluorouracil, mitomycin C, bleomycin, cisplatin, methotrexate have been tried in the neoadjuvant or the adjuvant setting, with limited success only.[3] The role of radiotherapy remains controversial with no definitive evidence.[3] On account of the rarity of the disorder, there are no large studies till date to enable an evidence based therapeutic approach.

Troublesome recurrences of BLT frequently occur , with the mean period to relapse having been reported as 10 months, by Chu et al.[3] Optimal management strategies both in the primary as well as recurrent setting thus still remain a matter of concern.

Reports in literature have been scarce with the largest series having been published in 1994 by Chu et al. of 42 cases.[3]

The inguinal region or the groin, till date has not been reported in literature as a site for BLT and to the best of our knowledge, this may be an index case in itself.

CONCLUSION

Buschke–Lowenstein tumor though rare in itself, may also present at unconventional sites such as the inguinal region. Early identification and prompt institution of adequate surgical management, with periodic follow‑ups form the mainstay of current therapy for this disease entity. This is what is to be borne in mind, for “what the mind knows is what the eyes see.”

REFERENCES1. Buschke A. Condyloma acuminata. In: Neisser A, editor. Stereoskopischer

Medizinischer Atlas. New York: Fischer; 1896. p. 8.2. Gormley RH, Kovarik CL. Human papillomavirus‑related genital disease in

the immunocompromised host: Part II. J Am Acad Dermatol 2012;66:883.e1‑17.

3. Chu QD, Vezeridis MP, Libbey NP, Wanebo HJ. Giant condyloma acuminatum (Buschke‑Lowenstein tumor) of the anorectal and perianal regions. Analysis of 42 cases. Dis Colon Rectum 1994;37:950‑7.

4. Sterry W, Paus R, Burgdorf W. Dermatology. Thiemes Clinical Companions. Acta Dermatovenerol Croat 2008;16:172.

5. Bertram P, Treutner KH, Rübben A, Hauptmann S, Schumpelick V. Invasive squamous‑cell carcinoma in giant anorectal condyloma (Buschke‑Löwenstein tumor). Langenbecks Arch Chir 1995;380:115‑8.

Figure 1: Buschke–Lowenstein tumor of the inguinal region (gross)

Figure 2: Postoperative appearance after wide local excision

Figure 3: Microscopic appearance of the Buschke–Lowenstein tumor, via (H and E, ×1000)

Aggarwal and Aggarwal: Buschke-Lowenstein tumour of the inguinal region

109 Oncology, Gastroenterology and Hepatology Reports| Jul-Dec 2015 | Vol 4 | Issue 2

How to cite this article: Aggarwal P, Aggarwal G. Buschke‑Lowenstein tumor of the inguinal region: A rare entity in a rarer location. Onc Gas Hep Rep 2015;4:107‑9.Source of Support: Nil, Conflict of Interest: None declared.

6. Trombetta LJ, Place RJ. Giant condyloma acuminatum of the anorectum: Trends in epidemiology and management: Report of a case and review of the literature. Dis Colon Rectum 2001;44:1878‑86.

7. De Toma G, Cavallaro G, Bitonti A, Polistena A, Onesti MG, Scuderi N. Surgical management of perianal giant condyloma acuminatum (Buschke‑Löwenstein tumor). Report of three cases. Eur Surg Res 2006;38:418‑22.

8. Lee SH, McGregor DH, Kuziez MN. Malignant transformation of perianal

condyloma acuminatum: A case report with review of the literature. Dis Colon Rectum 1981;24:462‑7.