Squamous-cell Carcinoma of the Anus Arising in a Giant Condyloma Acuminatum: Report of a Case* JAMES T. STUaM, M.D., CARL E. CnmSTENSON, M.D., JOHN H. UECk~n, M.D., JOHN F. PEARY, Ja., M.D. Frora the Department ol Stogery, St. Paul-Ramsey Hospital, and the Division of Colon and Rectal Surgery, University of Mim,esola Health Sciences Center, Minneapolis, Minnesota

a meningomyelocele with neurologic i n v o l v e m e n t manifested by a neurogenic bladder a n d m i l d lower*extremity weakness. Six years prior to admission, t h e patient h a d s o u g h t t r e a t m e n t in a n o t h e r hospital for an ulcerating lesion of t h e left buttock located between the anus a n d ischial tuberosity. Biopsy h a d proved the lesion to be benign, a n d local t r e a t m e n t h a d been administered. D u r i n g the e n s u i n g six years a slow-growing mass developed in the perianal skin a n d left buttock, eventually m e a s u r i n g 10.0 • 5.0 cm in greatest dimensions. Nine m o n t h s prior to admission, the patient h a d again s o u g h t t r e a t m e n t , for an ulcerating lesion located w i t h i n the large mass on the left buttock. Biopsy showed squamous-cell carcinoma, a n d a 6.0-cm ellipse of skin was excised. T h e m a r g i n s of resection were free of tumor. A split-thickness skin graft placed over the defect failed to take. T h e patient was treated with sitz baths, a n d the defect r e m a i n e d clean b u t did n o t heal. Nine m o n t h s later, at the time of admission to this hospital, the p a t i e n t gave a two-week history of profuse rectal bleeding. P e r t i n e n t physical findings included pallor, a 10.0 • 5.0-cm mass of the left buttock a n d perianal skin, a n d a 6.0-cm d e n u d e d area w i t h i n this mass (Fig. 1). An ileal u r i n a r y conduit was present on the r i g h t a b d o m i n a l wall. H e m o g l o b i n was 5.7 g/100 ml. Results of b a r i u m e n e m a studies, p y e l o ~ a p h y , cystoscopy, a n d sigmoidoscopy were n o r m a l . T h e site of rectal bleeding was d e t e r m i n e d to be the anal portion of the t u m o r mass. Biopsy of the perianal skin showed squamous-cell carcinoma. Following transfusion to correct thee h e m o g l o b i n to I1 g/100 ml, a b d o m i n o p e r i n e a l resection, cystectomy, a n d wide excision of the mass in the buttock was carried out. T h e extensive procedure was necessary because of m u l t i p l e fistulous tracts e x t e n d i n g widely in the buttocks a n d invasion of t u m o r into the pelvis. T h e 20.0 • 20.0-cm perineal w o u n d was left open (Fig. 2). Microscopic sections of the definitive t u m o r resection showed the edges of the resection to be

CONDYLOMA ACUMINATUM is a benign

disease caused by a virus that affects principally the genital and perianal areas. Although the lesions can recur following treatment, they are not invasive and usually are responsive to local therapeutic modalities. Rarely, a giant condyloma that invades contiguous structures occurs. This form of the disease was first described by Buschke in t896 and elaborated upon by Buschke and Loewenstein I in 1925. T h e terms "Buschke-Lowenstein tumor,', "giant condyloma acuminatum," and "carcinomalike condyloma" are synonymous. T h e giant condyloma acuminatum usually occurs on the penis. Only [our of approximately 60 reported patients with giant condylomas have had primary anorectal lesions. None of these four patients had a malignancy associated with the giant condyloma. This report describes a patient with a perianal giant condylorna who was found to have a simultaneous squamous-cell carcinoma of the anus. Report

of a Case

A 49-year-old m a n was admitted to the hospital on May 3, 1973. T h e patient h a d been born w i t h * Received for publication July 24, 1974. Supported in part by the Saint P a u l - R a m s e y Hospital Medical E d u c a t i o n and Research F o u n d a tion. Address reprint requests to: Dr. Perry, Departm e n t of Surgery, St. P a u l - R a m s e y Hospital, 640 Jackson Street, St. Paul, Minnesota 55101.

147 Dis. Col. & Rect. March 1975

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Fro. 1. iilass.

Dis. Col. &Rect. March 1975

Giant condyloma, patient lying on right side. a, scrotum; b, perianal skin; c, t u m o r

Fro. 2.

PerineaI w o u n d at operation.

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FIG. 3.

SQUAMOUS-CELL CARCINOMA

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Microscopic section of tumor, T h e center of the picture shows s q u a m o u s - c e l l carcinoma.

covered by n o r m a l skin. T h e r e was an a b r u p t transition from n o r m a l skin to a raised lesion m a n i f e s t i n g severe hyperkeratosis and acanthosis, w i t h moderate focal papillomatosis. T h e acanthotic epidermis showed a markedly thickened ga-anular-cell layer. T h e epidermis t h r o u g h o u t the edges of this lesion d e m o n s t r a t e d excellent m a t u r a tion of cells from basal to superficial layer. Rare dyskeratotic cells a n d rare mitotic figures were seen. T o w a r d the central portion of the lesion there were solid masses a n d cords of s q u a m o u s cells r e a c h i n g into the u n d e r l y i n g dermis. M a n y of the larger masses contained central cystic degeneration, with severe dyskeratosis, large n u m b e r s of mitotic figures, a n d occasional atypical mitoses. In several areas the cells showed a marked loss of m a t u r a t i o n from the basal to the superficial layer. In addition, there was severe nuclear p l e o m o r p h i s m , with an increased nuclear cytoplasmic ratio a n d excessive basophilia. In s u m m a r y , the edges of this lesion h a d features of a r a t h e r classic giant condyloma with sinus tract f o r m a t i o n within the u n d e r l y i n g dermis a n d s u b c u t a n e o u s tissue. However, w i t h i n the central portion of this lesion there was invasive squamous-cell carcinoma (Fig. 3).

Eight h o u r s after operation the patient was ret u r n e d to the o p e r a t i n g r o o m because of h e m o r rhage from the perineal w o u n d . Multiple bleeding points were cauterized a n d ligated. Subsequently the patient m a d e a slow b u t u n c o m p l i c a t e d recovery. T h e p a t i e n t u n d e r w e n t staged d o s u r e of his large perineal w o u n d . A m o n t h after a b d o m i n o perineal resection, two skin flaps were mobilized for partial closure of the defect. Skin coverage of the buttocks was complete d a m o n t h later by split-thickness grafting (Fig. 4). T h e patient was discharged ten weeks after a b d o m i n o p e r i n e a l resection a n d did well w i t h o u t recurrence for seven m o n t h s . Multiple loci of r e c u r r e n t t u m o r a p p e a r e d over the left ischial area a n d in the perineal w o u n d eight m o n t h s postoperatively. Because o[ its location, the t u m o r could not be removed by surgical means, nor was the area o[ recurrence suitable for radiotherapy. A l t h o u g h the long-term outlook is poor, local f u l g u r a t i o n a n d topical 5-fluorouracil applications have controlled the t u m o r to date (one year following a b d o m i n o p e r i n e a l resection, at the time of this writing) a n d the patient is gainfully employed.

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FiG. 4. One m o n t h postoperatively the patient has skin coverage over the buttocks, but the perineal w o u n d remains open.

Discussion T h e histologic distinctions between condyloma acuminatum and Buschke-Loewenstein tumor are largely a matter of degree. T h e tumor shows marked papillary proliferation and acanthosis. This tumor infiltrates adjacent structures and burrows into them, forming numerous fistulous tracts. Neither local lymph node metastases nor distant hematogenous metastases have been reported. Giant condylomas occur most commonly on the external genitalia. Shah and Hertz s reported the two cases of female patients who had extensive anorectal giant condylomas. One patient underwent abdominoperineal resection but ultimately died as a result of massive hemorrhage

Dis. CoI.& Rect. i~larch 1975

from extensive recurrence. T h e second patient refused surgery and was not benefited by a course of radiotherapy. Knoblich and Failing4 reported one male patient who died with a massive anorectal giant condyloma. Judge 3 described a female patient with Buschke-Loewenstein tumor of the anorectum who was free of recurrence a year after abdominoperineal resection. Although Friedberg and Serlin 2 and Siegel6 have each reported a patient with squamous-cell carcinoma arising in anorectal condyloma acuminatum, none of the four previously reported patients with giant condylomas of the anorectum have had associated malignancy. T h e squamouscell carcinoma in our patient appears to have arisen d e n o v o within the giant condyloma. On the basis of experience with our patient and the published experience of others, radical surgical treatment of giant condyloma of the anorectum seems justified. T h e invasive nature of this tumor causes multiple fistulous tracts that may invade fascia, muscle, or rectum, and may cause inflammation, infection, and hemorrhage. Surgery may result in cure if performed early, and if margins of resection are liberal. T h r e e conclusions concerning anorectal giant condylomas can be drawn from the course of this patient: 1) the presence of Buschke-Loewenstein tumor does not rule out malignancy, and multiple biopsies should be taken; 2) satisfactory results may be obtained with abdominoperineal resection of this tumor; 3) anything less than radical removal of the tumor is inadequate treatment.

Summary Giant condyloma acuminatum (BuschkeLoewenstein tumor) is distinguished from condyloma acuminatum by its invasive na-

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t u r e a n d t h e f o r m a t i o n of m u l t i p l e f i s t u l o u s tracts. The Buschke-Loewenstein tumor u s u a l l y o c c u r s o n t h e penis, b u t f o u r patients with anorectal giant condylomas have been reported in medical literature. This is a r e p o r t of a p a t i e n t in w h o m a squamous-cell carcinoma developed within a p e r i a n a l g i a n t c o n d y l o m a ; h e r e c e i v e d satisf a c t o r y p a l l i a t i v e t r e a t m e n t by a b d o m i n o p e r i n e a l r e s e c t i o n a n d s u b s e q u e n t local treatment. Acknowledgment This paper was prepared with the technical assistance of Mr. Herbert Crandall.

15 l References

1. Buschke A, Loewenstein L: fAber Carcinomtihnliche Condylomata Accuminata des Penis. Klin Wochenschr 4: 1726, 1925 2. Friedberg MJ, Serlin O: Condyloma acuminarum: Its association with malignancy. Dis Colon Rectum 6: 352, 1963 3. Judge JR: Giant condyloma acuminatum involving vulva and rectum. Arch Pathol 88: 46, 1969 4. Knoblich R, Failing JF Jr: Giant condyloma acuminatum (Buschke-Lowenstein tumor) of the rectum. Am J Clin Pathol 48: 389, 1967 5. Shah IC, Hertz RE: Giant condyloma acuminature of the anorectum: Report of two cases. Dis Colon Rectum 15: 207, 1972 6. Siegel A: Malignant transformation of condyloma acuminatum: Review of the literature and report of a case. Am J Surg 103: 613, 1962

Announcement VIth. Latin American Association of Proctology Organized by the Latin American Association of Proctology (A.L.A.P.) President: Dr. Alberto Laurence Date: October 26th to 31st, 1975 Place: Buenos Aires, Argentina Scientific Program: will include simultaneous colloquiums, two television forums, lectures, a course on pathology of the anus, colon and rectum. Free communications. Simultaneous interpretations: Spanish-English, English-Spanish Information may be obtained from the Secretariat, Dr. A. Heidenreich, Av. Roque S,'ienz Pefia 1110, 2 ~ piso, Buenos Aires, Argentina.

Squamous-cell carcinoma of the anus arising in a giant condyloma acuminatum: report of a case.

Squamous-cell Carcinoma of the Anus Arising in a Giant Condyloma Acuminatum: Report of a Case* JAMES T. STUaM, M.D., CARL E. CnmSTENSON, M.D., JOHN H...
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