Anal / Homosexual Sex Issues. A surgical approach to giant condyloma (Buschke-Löwenstein tumour) with underlying superficial vulvar carcinoma: A case report

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Authors: Josko Zekan Davor Petrovic Samer El-Safadi Maja Banovic
Davor Hulina Zlatko Hrgovic
Corresponding author: Samer El-Safadi [ samer.el-
saf...@gyn.med.uni-giessen.de ]
View Affiliations

Published online on: Wednesday, November 14, 2012
Pages: 541-543 DOI: 10.3892/ol.2012.1027

Abstract

Anogenital warts (condyloma acuminatum or venereal warts) are a
common sexually transmitted disease in males and females. Common
clinical treatment of anogenital warts is conservative, however,
in extreme cases conservative therapy is insufficient and
surgical excision is required. Giant condyloma acuminata
(Buschke-Löwenstein tumour) is an extremely rare clinical type
of genital wart, characterised by aggressive down growth into
underlying dermal structures. A 55-year-old female presented
with cauliflower-like growth over the anogenital and sacral
region, earlier diagnosed as condyloma acuminatum which was
resistant to conservative therapy. During the period between
2005 and 2008 the patient underwent five surgical procedures.
Due to the size and location of the tumour, gynaecological and
plastic surgeons were involved in the procedures. In addition,
definitive histology examination identified a superficial vulvar
carcinoma.
Introduction

Anogenital warts (condyloma acuminatum or venereal warts) are a
common sexually transmitted disease among females and males
(1,2). The causal role of human papillomaviruses (HPV) in
anogenital wart formation has been firmly established
biologically and epidemiologically (3,4). Genital HPV infections
are transmitted primarily through sexual contact, with a
lifetime risk of 50–80% (5). The highest rate of genital HPV
infection has been identifed in adults between 18 and 28 years
of age (6,7). The immune system effectively repels the majority
of HPV infections and is associated with marked localised cell
mediated immune responses. However, approximately 10% of
individuals develop a persistent infection, with risk of
developing benign proliferative lesions, high-grade precursors
and eventually invasive carcinomas (8). HPVs are classified into
high- or low-risk types depending on oncogenic potential. Low-
risk types 6 and 11 are isolated in approximately 90% of genital
wart cases (3). The most common clinical treatment is
conservative, with local chemical or physical destruction and
immunological therapy (9). In more extreme cases conservative
therapy is insufficient and surgical excision is required.

Giant condyloma acuminata (GCA; Buschke-Löwenstein tumour) is an
extremely rare clinical form of genital warts, characterised by
aggressive down growth into underlying dermal structures
(10,11). A complex histological pattern may exist with areas of
benign condyloma intermixed with foci of atypical epithelial
cells or well differentiated squamous cell carcinoma. GCA is
mainly localised to the genital region, however, in rare cases
the tumour is localised to distinct histological zones of the
anorectal region. Due to infiltration of the underlying tissue,
fistulae and abscesses may be observed. GCA is resistant to
chemotherapy or radiotherapy and usually requires local radical
resection for curative treatment. The study was approved by the
ethics committee of University Medical School, Zagreb. Written
informed consent was obtained from the patient.

Case report

A 55-year-old female presented with cauliflower-like growth over
the anogenital and sacral region. The growth had been diagnosed
previously as condyloma acuminatum which was resistant to
conservative therapy (Fig. 1). The patient’s medical history was
as follows: at 17 years old the patient was diagnosed with
infective mononucleosis and 6 months later with viral pneumonia.
In 1979, the patient suffered from pyelonephritis caused by E.
coli, with subsequent unilateral permanent kidney lesion.
Multiple condyloma were diagnosed for the first time during the
patient’s first pregnancy in 1970, at the perineal surface and
were surgically removed in the same year, following delivery.
Four years following excision recurrence was identified and was
treated successfully with albothyl solution for two months.


Figure 1.
Patient with giant condyloma prior to reconstructive surgery.

During the first trimester of the patient’s second pregnancy
(1978) warts appeared for the third time with altered clinical
presentation; spread across the entire anogenital region
(perineum, anal orifice and labia majora) and became
multilayered and painful. Despite repeated albothyl therapy,
growth continued. Prior to labour the warts were removed by
electro-cauterisation and the whole surface was treated locally
with interferon (IFN) ointment. During the following year there
were no visible warts. Between 1980 and 1981, due to recurrence,
the patient underwent two additional surgical procedures
followed by IFN treatment. Positive response to treatment lasted
for 6–8 months and was followed by five excision procedures
under local anaesthesia between 1983 and 1984. All condyloma
were removed. The severity of the disease increased the
following year and was successfully treated with IFN ointment
over one year. In 1986, tumour size increased again. The patient
recieved local IFN therapy, however, treatment response was
inadequate as the tumour was reduced in size by 50%. Condyloma
size remained constant until 2003 when the patient entered
menopause. Podophyllin treatment was administered, however, the
side-effects included bilateral inguinal lymphadenopathy and
marked pain. At this point the patient was admitted to our unit.

Between 2005 and 2008 the patient underwent five surgical
procedures. The procedures were performed by gynaecological and
plastic surgeons due to the size and location of the tumour. The
first surgery was a loop colostomy on the sigmoid colon
performed by an abdominal surgeon. The following procedures were
performed by a plastic and reconstructive surgeon over four
surgical periods; radical excision of the vulvar, perineal, anal
and sacral condyloma with the preservation of urethral orifice,
vagina and anus (Fig. 2). Reconstruction of the dorsal defect
was formed using two large fascio-cutaneal flaps based on the
superior gluteal artery. The remaining defect of the vulva was
reconstructed by local transpositional fascio-cutaneal flaps
from the medial side of the upper thighs. Following the third
surgery, a postoperative infection developed with partial
dehiscence of the local transpositional flaps from the upper
thighs, therefore, necrectomy was performed, covering the
residual with a split thickness skin graft from the right upper
thigh. Seven months following, excision of the cicatrices was
performed due to development of contractures of the perineal
skin and the defects split thickness skin graft was utilised for
covering. The final surgery was the occlusion of the colostomy
performed by the abdominal surgeon (Fig. 3).


Figure 2:.
Patient during the surgical proceedure.


Figure 3:.
Patient directly following surgery.

Following 6 months, local status was a preserved urethral
orifice and vaginal introitus with a small rectal mucosa
prolapse (Fig. 4).


Figure 4.
Patient following the healing process.

In the present case, the giant condyloma appeared to exhibit
characteristics of a normal condyloma acuminatum and a
superficial planocellular carcinoma. The majority of the
material received for pathology analysis was condyloma
accuminatum with prominent acanthosis, dyskeratosis, hyper-
keratosis and prominent granular layer. In superficial
epithelial cells typical perinuclear cytoplasmic ‘halos’ and
pyknotic or slightly enlarged nuclei were observed and bi-
nuclear cells were present. In specific areas invasive
superficial squamous carcinoma was identified with invasion of
the underlaying dermis with small clusters of cells accompanied
with prominent mononuclear inflammatory infiltrate.
Immunohistochemical analysis with p16 monoclonal antibody (clone
E6H4 against p16 protein; CINtec Histology; Roche Diagnostics
GmbH, Mannheim, Germany) revealed positivity in tumour cells.
Additional analysis using Digene Hybrid Capture 2 (Qiagen,
Hilden, Germany) was performed and the presence of HPV genotype
6 and 11 was confirmed (Fig. 5).


Figure 5.
Histology of the giant condyloma.

Discussion

Anogenital warts are the most common outcome of HPV genital
infection. Therapeutics against this sexually transmitted
disease are currently associated with low efficacy, due to a
30–70% recurrence rate identified six months following therapy
administration (9). In rare cases, anogenital warts develop into
extremely large tumour masses leading to deterioration of
patient quality of life. An identified underlying histopathology
of specific cases of giant condyloma is superficial
planocellular carcinoma. Patients with high susceptibility to
local development and fast progression (in growth and
malignancy) and the highest rate of recurrence often exhibit
various types of immunodeficiency. In addition, immunodeficiency
leads to difficulties in evaluation of optimal therapeutic
management. However, patients with no marked immunodeficiency
and treatment-resistent genital warts have been identified.
Furthermore, condyloma lesions occasionally form large exophytic
masses, interfering with intercourse, normal urination,
defecation or vaginal delivery.

Commonly, GCA develops as cauliflower-like masses and the
tumours exhibit histological features of pseudo-epitheliomatous
profileration and local invasion. In the absence of metastases,
they are termed Buschke-Löwenstein tumours. Due to the
aggressive local development of these masses they belong to the
verrucous carcinoma group, although a malignant histological
alteration in the form of micro-invasive carcinoma or well-
differentiated epidermoid keratinising carcinoma has been
reported. Due to the high frequency of local recurrence, radical
surgical excision is the current treatment of choice as topical
preparations and chemotherapy are generally considered
ineffective (12). The method selected for reconstruction is
crucial, particularly in neglected cases similar to the present
case study. Local tissue availability and the patient’s
condition and attitude towards the development of the disease
are major factors for reconstruction with local fascio-cutaneous
flaps. Five years following surgury the present patient is
disease-free with no recurrence.

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