EP310 - A Buschke-Löwenstein Tumor with Malignant Transformation Diagnosed After Wide Local Excision

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Purpose/Background: We present an interesting case of a Buschke-Löwenstein tumor (BLT) with atypical malignant transformation following wide local excision with immediate soft tissue reconstruction. The patient is a 54 year-old male with a past medical history significant for hemorrhoids and alcohol use disorder. The patient presented with rectal pain and associated rectal bleeding. On physical exam, he was found to have a ~15x8cm fungating, friable, mobile, perianal condylomatous mass with contact bleeding surrounding 70-80% circumference of anus, consistent with a giant anal condyloma (Figure 1A-B). Anoscopy was performed in office, revealing the giant perianal condyloma, with normal rectal tone and minimal involvement of the anal canal or concern for sphincter involvement. A computed tomography (CT) of the abdomen and pelvis revealed a large soft tissue abnormality adjacent to the posterior aspect of anal canal, extending to the midline buttocks region.

Methods/Interventions: Methods/Interventions
Following multidisciplinary discussion, we elected for fecal diversion to allow for wound healing once definitive management was performed, which would likely require soft tissue reconstruction. The patient subsequently underwent laparoscopic loop ileostomy creation and biopsy of the mass. A colonoscopy was also performed, which did not show any other concerning lesions. Final pathology returned as a well-differentiated squamous lesion consistent with keratoacanthoma. In light of these findings, the patient was taken to the operating room 6 weeks later for definitive management. Wide local excision was performed, followed by bilateral V-Y fasciocutaneous advancement flaps by Plastic Surgery to reconstruct perianal area (Figure 1C-1F).

Results/Outcomes: Final pathology revealed invasive, well-differentiated squamous cell carcinoma (SCC) with features of verrucous carcinoma (VC). Resection margins were free of invasive carcinoma or dysplasia. Human papilloma virus (HPV) in-situ hybridization (ISH) testing for both high risk and low risk genotypes were negative. The patient healed well (Figure 1G-H). Given the HPV ISH results, a definitive diagnosis of VC was made. The patient was discussed at multidisciplinary tumor board and decision for further imaging and observation versus adjuvant therapy was discussed.

Conclusion/Discussion: Giant anal condyloma acuminatum or BLTs have an estimated incidence of 0.1%. They are typically associated with sexually-transmitted HPV infection. Although usually histologically benign at diagnosis, they are clinically destructive, cauliflower-like fungating lesions that are locally invasive, with a high incidence of recurrence if not managed properly. Up to 50% of BLTs may undergo malignant transformation into SCC. While true giant condyloma with SCC transformation is HPV-induced (usually HPV 6,11), VC, which is a varient of SCC, is not virally induced. Therefore, HPV ISH allowed for definitive diagnosis in this case. Mainstay treatment for BLTs and VC involves upfront surgical resection with wide-local excision. The use of chemoradiotherapy is not as well understood, however has been used, particularly in cases with positive margins and/or involvement of perianal structures such as the anal sphincter complex. Given the different way that HPV-associated SCC of the anus is treated, diagnosis and histologic differentiation from VC is essential. We hope this case provides interesting insight into the spectrum of disease of BLTs and management approaches.