© 1991 Kargcr AG, Basel 0011-9075/91/1823-0193 $2.75/0
Dermatológica 1991;182:193-195
Verrucous Carcinoma of a Leg Amputation Stump R.A. Schwartz, M.P. Bagley, C.K. Janniger, W. C. Lambert Dermatology, UMDNJ-New Jersey Medical School, Newark, NJ; Dermatology, East Orange Veterans Administration Medical Center. East Orange, NJ. USA
Key Words. Verrucous carcinoma • Cutaneous squamous cell carcinoma • Skin neoplasms Abstract. Verrucous carcinoma of the skin is a rare cancer which tends to appear on the soles of the feet, although it can appear elsewhere on the cutaneous surface. It has not, to our knowledge, been previously reported on a leg ampu tation stump.
Case Report A 65-year-old white man was referred for the evaluation of a tumor of his right leg stump of 9 months’ duration. The lesion had begun as an ulcer approximately 2 years previously and had been treated with local wound care. It had increased in size for 3 months prior to our evalua tion. By this time the patient noted a foul-smelling discharge from the site. He had suffered a traumatic amputation of the right leg in 1942 with a resultant below-thc-kncc amputation. A leg prosthesis had been worn since that time.
On physical examination there was an oval skin-colored verru cous tumor about 5 cm in diameter on the right leg stump with a foul smelling discharge (fig. 1). A chest X-ray was within normal limits. A gallium scan demonstrated no bony involvement. A skin biopsy was performed followed by a local excision with a revision of the amputa tion. Histologic examination showed a massively exophytic hyperplastic epidermis. Marked hyperkeratosis was evident, with a prominent gran ular layer. The individual kératinocytes appeared very bland cytologically. Mitotic figures, individual cell necrosis and multinucleated kéra tinocytes were not evident. The tumor projected deeply into the dermis as bluntly shaped masses (fig. 2). Beneath the tumor was a marked infil trate of lymphohistiocytic origin. Attempts to stain the specimen for human papillomaviruses were not successful, due to processing of the specimen with picric acid.
Discussion The biologic behavior of VC is intriguing [1-3]. It is an unusual type of low-grade squamous cell carcinoma which clinically appears as a slowly enlarging warty mass which is both exophytic and endophytic, extending deeply into the tissues locally. Histologically, it is a well-differentiated squamous cell carcinoma which displays local invasion and very little if any dysplasia [6,7]. VC has certain char acteristic sites of predilection, such as the sole of the foot, where it was named epithelioma cuniculatum because of its resemblance histologically to multiple crypts of a rabbit burrow [1, 2], The condyloma-acuminatum-like carci-
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The verrucous carcinoma (VC) is best viewed as a very low-grade and well-differentiated carcinomatous process which displays a marked tendency to compress and dis place deeper tissues by downward growth rather than infiltrating them or by metastasizing! 1.2]. In 1948 Acker man [3] described similar neoplasms of the oral cavity in 31 patients, invoking the term verrucous carcinoma to denote this concept of a locally aggressive exophytic lowgrade squamous cell carcinoma of very little metastatic potential. The VC has also been described involving a pilonidal sinus [4] and on many other sites, most typically on the soles of the feet, oral cavity and genitalia. How ever, this is the second patient to our knowledge in whom the VC appeared on an amputation stump, the first being a patient with a partial traumatic amputation of the foot 20 years preceding the tumor [5]. Our patient is the first who developed VC on a leg amputation stump.
194
Schwartz/Bagley/Janniger/Lambert
noma of the penis [8] described by Buschke and Lovvenstcin in 1925 was a VC [9, 10). It also occurs perianally, on the vagina and may rarely be seen anywhere on the body including the face, buttocks, palms or fingers [5, 11-17). Other names for VC employed include papillomatosis cutis carcinoides, papillomatosis cutis and VC of the oral cavity (2). This tumor occurs predominately in middle-aged men, although it can occur in both sexes and in a wide age range (23-85 years). Etiologically, it is most likely related to a human papillomavirus infection and may also be associ ated with chemical carcinogens or cocarcinogens [1, 5, 12). VC may develop at the site of long-standing cuta neous scars [5. 18-20]. Clinically, this tumor may become
Fig. 2. The tumor projecting into the dermis as blunt masses. Hematoxylin-eosin. Original magnification x78.
quite bulky, as it enlarges with extensive compression of underlying tissues. It may produce a large foul-smelling mass with the consistency of an overripe orange. Eventu ally, it will penetrate underlying tissue and bone. Metastases of this well-differentiated cancer are rare, although in long-standing or irradiated tumors spread to the regional lymph nodes has occurred [21-26]. This is to our knowledge the first patient observed with VC of the leg amputation stump and the second with VC within an amputation stump. The factors which might lead to the development of VC at this scar site are unclear. However, it appeared on the most acral portion of the lower extremity in our patient, making it comparable to the epithelioma cuniculatum.
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Fig. 1. VC of the stump scar.
195
Verrucous Carcinoma
1 Schwartz RA: Squamous cell carcinoma; in Schwartz R A (eil): Skin Cancer: Recognition and Management. New York. Springer. 1988. pp 36—47. 2 Schwartz RA: Verrucous carcinoma of the skin; in Demis DJ (ed): Clinical Dermatology, ed 18. Philadelphia. Harperand Row. 1991. unit 21-22, pp 1-6. 3 Ackerman LV: Verrucous carcinoma of the oral cavity. Surgery 1948;23:670-678. 4 Norris CS: Giant condyloma acuminatum (Buschke-Lowenstein tumor) involving a pilonidal sinus: A case report and review of the literature. J Surg Oncol 1983;22:47-50. 5 Kao GF, Graham JH. Helwig EB: Carcinomacuniculatum (verru cous carcinoma of the skin). A clinicopathologic study of 46 cases with ultrastructural observations. Cancer 1982;49:2395-2403. 6 Wick MR. Manivel JC. Millns JL: Histopathologic considerations in the management of skin cancer: in Schwartz RA (ed): Skin Can cer: Recognition and Management. New York. Springer. 1988. pp 246-275. 7 Priolcau PG. Santa Cruz DJ. Meyer JS. Bauer WC: Verrucous car cinoma. A light and electron microscopic, autoradiographic and immunofluorescence study. Cancer 1980:45:2849-2857. 8 Löwenstein LW: Carcinoma-like condylomata acuminata of the penis. Med Clin North Am 1939:23:789-795. 9 Jablonska S. Schwartz RA: Buschkc-Loewcnstcin tumor: in Demis DJ (ed): Clinical Dermatology, ed. 18. Philadelphia, Harper and Row. Unit 14-15. 1991. pp 1-6. 10 Schwartz RA: Giant condyloma acuminatum of Buschke and Loewenstein. J Am Acad Dermatol. 1990:23:723-727. 11 BendelacA.GrossinM. Sigal M .étal: L'épithéliomacuniculatum. Ann Pathol 1984:4:223-229. 12 Knobler RM. Schneider S, Neumann RA et al: DNA dot-blot hybridization implicates human papillomavirus type 11-DNA in epithelioma cuniculatum. J Med Virol 1989:29:33-37. 13 Balázs M: Buschke-Loewenstein tumour. A histologic and ultrastructural study of six cases. Virchows Arch (A) 1986;410:83-92. 14 Kumar AS. George E. Pandhi RK: Epithelioma cuniculatum pal mare (verrucous carcinoma) with palmo-plantar keratoderma. Indian J Dermatol Venereol Leprol 1984;50:269-270. 15 Nguyen KO. McMarlin SL: Verrucous carcinoma of the face. Arch Dermatol 1984;120:383-385.
16 Coldiron BM. Brown FC. Freeman RG: Epithelioma cuniculatum
17 18
19
20
21
22
23
24 25
26
(carcinoma cuniculatum) of the thumb: A case report and literature review. J Dermatol Surg Oncol 1986;12:1150-1155. Collison DW. Mikhail GR: Carcinoma cuniculatum of the hand. Arch Dermatol 1989;125:1335-1336. Mayron R. Grimwood RE. Siegle RJ, Camisa C: Verrucous carci noma arising in ulcerative lichen planus of the soles. J Dermatol Surg Oncol 1988;14:547-551. Klima M. Kurds B. Jordan PI I Jr: Verrucous carcinoma of the skin. J Cutan Pathol 1980:7:88-98. Turner JE. Hodge SJ. Callcn JP: Verrucous carcinoma in a renal transplant patient alter long-term immunosuppression. Arch Der matol 1980:116:1074-1076. Perez CA. Kraus FT. Evans JC. et al: Anaplastic transformation in verrucous carcinoma of the oral cavity after radiation therapy. Radiology 1966;86:108-115. Dcmian SDE. Bushkin FL. Echevarria RA: Perineural invasion and anaplastic transformation of verrucous carcinoma. Cancer 1973:32:395-401. Nair MK. Sankaranarayanan R. Padmanabhan TK. Madhu CS: Oral verrucous carcinoma. Treatment with radiotherapy. Cancer 1988:61:458—461. Owen WR. Wolfe ID. Burnett JW. et al: Epithelioma cuniculatum. South Med J 1978:71:477-479. Reingold IM. Smith BR. Graham JH: Epithelioma cuniculatum pedis, a variant of squamous cell carcinoma. Am J Clin Pathol 1978:69:561-565. McKee PH. Wilkinson JD. Black MM, et al: Carcinoma (epithe lioma) cuniculatum: A clinico-pathological study of nineteen cases and review' of the literature. Histopathology 1981;5:425-436.
Received: July 13. 1990 Accepted: August 31. 1990 Prof. R.A. Schwartz Dermatology New Jersey Medical School 185 South Orange Avenue Newark. NJ 07103-2714 (USA)
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References
Dermatológica 1991;182:193-195
Verrucous Carcinoma of a Leg Amputation Stump R.A. Schwartz, M.P. Bagley, C.K. Janniger, W. C. Lambert Dermatology, UMDNJ-New Jersey Medical School, Newark, NJ; Dermatology, East Orange Veterans Administration Medical Center. East Orange, NJ. USA
Key Words. Verrucous carcinoma • Cutaneous squamous cell carcinoma • Skin neoplasms Abstract. Verrucous carcinoma of the skin is a rare cancer which tends to appear on the soles of the feet, although it can appear elsewhere on the cutaneous surface. It has not, to our knowledge, been previously reported on a leg ampu tation stump.
Case Report A 65-year-old white man was referred for the evaluation of a tumor of his right leg stump of 9 months’ duration. The lesion had begun as an ulcer approximately 2 years previously and had been treated with local wound care. It had increased in size for 3 months prior to our evalua tion. By this time the patient noted a foul-smelling discharge from the site. He had suffered a traumatic amputation of the right leg in 1942 with a resultant below-thc-kncc amputation. A leg prosthesis had been worn since that time.
On physical examination there was an oval skin-colored verru cous tumor about 5 cm in diameter on the right leg stump with a foul smelling discharge (fig. 1). A chest X-ray was within normal limits. A gallium scan demonstrated no bony involvement. A skin biopsy was performed followed by a local excision with a revision of the amputa tion. Histologic examination showed a massively exophytic hyperplastic epidermis. Marked hyperkeratosis was evident, with a prominent gran ular layer. The individual kératinocytes appeared very bland cytologically. Mitotic figures, individual cell necrosis and multinucleated kéra tinocytes were not evident. The tumor projected deeply into the dermis as bluntly shaped masses (fig. 2). Beneath the tumor was a marked infil trate of lymphohistiocytic origin. Attempts to stain the specimen for human papillomaviruses were not successful, due to processing of the specimen with picric acid.
Discussion The biologic behavior of VC is intriguing [1-3]. It is an unusual type of low-grade squamous cell carcinoma which clinically appears as a slowly enlarging warty mass which is both exophytic and endophytic, extending deeply into the tissues locally. Histologically, it is a well-differentiated squamous cell carcinoma which displays local invasion and very little if any dysplasia [6,7]. VC has certain char acteristic sites of predilection, such as the sole of the foot, where it was named epithelioma cuniculatum because of its resemblance histologically to multiple crypts of a rabbit burrow [1, 2], The condyloma-acuminatum-like carci-
Downloaded by: Univ. of California Santa Barbara 128.111.121.42 - 2/24/2018 1:47:50 PM
The verrucous carcinoma (VC) is best viewed as a very low-grade and well-differentiated carcinomatous process which displays a marked tendency to compress and dis place deeper tissues by downward growth rather than infiltrating them or by metastasizing! 1.2]. In 1948 Acker man [3] described similar neoplasms of the oral cavity in 31 patients, invoking the term verrucous carcinoma to denote this concept of a locally aggressive exophytic lowgrade squamous cell carcinoma of very little metastatic potential. The VC has also been described involving a pilonidal sinus [4] and on many other sites, most typically on the soles of the feet, oral cavity and genitalia. How ever, this is the second patient to our knowledge in whom the VC appeared on an amputation stump, the first being a patient with a partial traumatic amputation of the foot 20 years preceding the tumor [5]. Our patient is the first who developed VC on a leg amputation stump.
194
Schwartz/Bagley/Janniger/Lambert
noma of the penis [8] described by Buschke and Lovvenstcin in 1925 was a VC [9, 10). It also occurs perianally, on the vagina and may rarely be seen anywhere on the body including the face, buttocks, palms or fingers [5, 11-17). Other names for VC employed include papillomatosis cutis carcinoides, papillomatosis cutis and VC of the oral cavity (2). This tumor occurs predominately in middle-aged men, although it can occur in both sexes and in a wide age range (23-85 years). Etiologically, it is most likely related to a human papillomavirus infection and may also be associ ated with chemical carcinogens or cocarcinogens [1, 5, 12). VC may develop at the site of long-standing cuta neous scars [5. 18-20]. Clinically, this tumor may become
Fig. 2. The tumor projecting into the dermis as blunt masses. Hematoxylin-eosin. Original magnification x78.
quite bulky, as it enlarges with extensive compression of underlying tissues. It may produce a large foul-smelling mass with the consistency of an overripe orange. Eventu ally, it will penetrate underlying tissue and bone. Metastases of this well-differentiated cancer are rare, although in long-standing or irradiated tumors spread to the regional lymph nodes has occurred [21-26]. This is to our knowledge the first patient observed with VC of the leg amputation stump and the second with VC within an amputation stump. The factors which might lead to the development of VC at this scar site are unclear. However, it appeared on the most acral portion of the lower extremity in our patient, making it comparable to the epithelioma cuniculatum.
Downloaded by: Univ. of California Santa Barbara 128.111.121.42 - 2/24/2018 1:47:50 PM
Fig. 1. VC of the stump scar.
195
Verrucous Carcinoma
1 Schwartz RA: Squamous cell carcinoma; in Schwartz R A (eil): Skin Cancer: Recognition and Management. New York. Springer. 1988. pp 36—47. 2 Schwartz RA: Verrucous carcinoma of the skin; in Demis DJ (ed): Clinical Dermatology, ed 18. Philadelphia. Harperand Row. 1991. unit 21-22, pp 1-6. 3 Ackerman LV: Verrucous carcinoma of the oral cavity. Surgery 1948;23:670-678. 4 Norris CS: Giant condyloma acuminatum (Buschke-Lowenstein tumor) involving a pilonidal sinus: A case report and review of the literature. J Surg Oncol 1983;22:47-50. 5 Kao GF, Graham JH. Helwig EB: Carcinomacuniculatum (verru cous carcinoma of the skin). A clinicopathologic study of 46 cases with ultrastructural observations. Cancer 1982;49:2395-2403. 6 Wick MR. Manivel JC. Millns JL: Histopathologic considerations in the management of skin cancer: in Schwartz RA (ed): Skin Can cer: Recognition and Management. New York. Springer. 1988. pp 246-275. 7 Priolcau PG. Santa Cruz DJ. Meyer JS. Bauer WC: Verrucous car cinoma. A light and electron microscopic, autoradiographic and immunofluorescence study. Cancer 1980:45:2849-2857. 8 Löwenstein LW: Carcinoma-like condylomata acuminata of the penis. Med Clin North Am 1939:23:789-795. 9 Jablonska S. Schwartz RA: Buschkc-Loewcnstcin tumor: in Demis DJ (ed): Clinical Dermatology, ed. 18. Philadelphia, Harper and Row. Unit 14-15. 1991. pp 1-6. 10 Schwartz RA: Giant condyloma acuminatum of Buschke and Loewenstein. J Am Acad Dermatol. 1990:23:723-727. 11 BendelacA.GrossinM. Sigal M .étal: L'épithéliomacuniculatum. Ann Pathol 1984:4:223-229. 12 Knobler RM. Schneider S, Neumann RA et al: DNA dot-blot hybridization implicates human papillomavirus type 11-DNA in epithelioma cuniculatum. J Med Virol 1989:29:33-37. 13 Balázs M: Buschke-Loewenstein tumour. A histologic and ultrastructural study of six cases. Virchows Arch (A) 1986;410:83-92. 14 Kumar AS. George E. Pandhi RK: Epithelioma cuniculatum pal mare (verrucous carcinoma) with palmo-plantar keratoderma. Indian J Dermatol Venereol Leprol 1984;50:269-270. 15 Nguyen KO. McMarlin SL: Verrucous carcinoma of the face. Arch Dermatol 1984;120:383-385.
16 Coldiron BM. Brown FC. Freeman RG: Epithelioma cuniculatum
17 18
19
20
21
22
23
24 25
26
(carcinoma cuniculatum) of the thumb: A case report and literature review. J Dermatol Surg Oncol 1986;12:1150-1155. Collison DW. Mikhail GR: Carcinoma cuniculatum of the hand. Arch Dermatol 1989;125:1335-1336. Mayron R. Grimwood RE. Siegle RJ, Camisa C: Verrucous carci noma arising in ulcerative lichen planus of the soles. J Dermatol Surg Oncol 1988;14:547-551. Klima M. Kurds B. Jordan PI I Jr: Verrucous carcinoma of the skin. J Cutan Pathol 1980:7:88-98. Turner JE. Hodge SJ. Callcn JP: Verrucous carcinoma in a renal transplant patient alter long-term immunosuppression. Arch Der matol 1980:116:1074-1076. Perez CA. Kraus FT. Evans JC. et al: Anaplastic transformation in verrucous carcinoma of the oral cavity after radiation therapy. Radiology 1966;86:108-115. Dcmian SDE. Bushkin FL. Echevarria RA: Perineural invasion and anaplastic transformation of verrucous carcinoma. Cancer 1973:32:395-401. Nair MK. Sankaranarayanan R. Padmanabhan TK. Madhu CS: Oral verrucous carcinoma. Treatment with radiotherapy. Cancer 1988:61:458—461. Owen WR. Wolfe ID. Burnett JW. et al: Epithelioma cuniculatum. South Med J 1978:71:477-479. Reingold IM. Smith BR. Graham JH: Epithelioma cuniculatum pedis, a variant of squamous cell carcinoma. Am J Clin Pathol 1978:69:561-565. McKee PH. Wilkinson JD. Black MM, et al: Carcinoma (epithe lioma) cuniculatum: A clinico-pathological study of nineteen cases and review' of the literature. Histopathology 1981;5:425-436.
Received: July 13. 1990 Accepted: August 31. 1990 Prof. R.A. Schwartz Dermatology New Jersey Medical School 185 South Orange Avenue Newark. NJ 07103-2714 (USA)
Downloaded by: Univ. of California Santa Barbara 128.111.121.42 - 2/24/2018 1:47:50 PM
References
