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Squamous cell carcinoma occurring in chronic perianal pyoderma
S Q U A M O U S C E L L C A R C I N O M A O C C U R R I N G IN CHRONIC PERIANAL PYODERMA By BORJE SUNDELL,M.D., and CARLM~LLER, M.D.
Department of Plastic Surgery, First Surgical Clinic, University Hospital, Helsinki, Finland SQUAMOVS cell carcinoma in chronic perianal pyoderma is a rare lesion. There are only a few reports on carcinoma arising secondary to ache conglobata (Hellier, I939 ; Schiff and Kern, I957; Dillon and Spjut, I964) and to anorectal fistulae (Rosser, i 9 3 i ; Rundle and Hales, x953 ; Schwarz, I963). Chronic inflammation in the perianal skin and subcutaneous tissue may continue for a long period without affecting the general health or the working capacity. Similar clinical features (M611er, Saksela and Sundell, I969) can be produced by ache conglobata, hidradenitis suppurativa, anal fistula: and pilonidal sinuses and the correct origin may be difficult to determine because of the inflammation (Jackman and McQuarrie, 1949). Carcinoma arising in an area of untreated or unhealed fistula: and draining abscesses permits early diagnosis by inspection and palpation. I f any area of hard induration is noted, especially if combined with ulceration of the skin, the suspicion of malignant change should be aroused and a biopsy done (Turell, I962). This is a report of two cases with uniform clinical appearance but with different histological background which developed squamous cell carcinoma after years of chronic inflammation. Case I.--A 52-year-old serviceman was first seen by us in February I967. Perianal pyoderma draining through his gluteal region developed in x94o. The fistulous tract was unroofed in I94I but never healed completely. In I964 multiple abscesses developed with sinuses draining a foul-smelling mixture of pus and blood over the gluteal region. He was seen in another hospital for conservative treatment at that time. In December I966 a ha~morrhage from the left buttock brought the patient to the II Surgical Clinic, University Hospital, Helsinki. The whole left gluteal region was infected and indurated (Fig. I, n). There were large, elevated fungating tumour masses on the upper part of the left buttock. On the medial side~ a large anorectal fistula was observed. Biopsy of the tumour showed a keratinous type of epidermoid carcinoma. Fismlography revealed a fistula immediately above the anal sphincter, directed towards the left buttock. On sigmoidoscopy, inflammation of the rectal mucosa was seen but no malignant disease could be observed. In February x967 a transverse colostomy was made ; the patient was then admitted to the Department of Plastic Surgery for further treatment of his left buttock. In March I967 a wide resection of skin, subcutaneous and muscular tissue including the tumour masses and the sinuses of the skin of the left buttock was performed. The whole tumour area was removed up to the anal sphincter which was left intact. The tissue defect extended over the whole left buttock and an area around the anal canal. The defect was immediately covered with a rotation flap from the lateral aspect of the buttock and partly with a medium split thickness skin graft from the thigh. The flap and the graft healed well. InApril x967 an abdomino-perineal resection was performed. Apermanent sigmoidostomy was left. No metastases could be found at that time, Carcinoma of the colon could not be verified ; only fibrosis and granulation tissue appeared in the histological specimens. In May I967 the patient was readmitted for suspected recurrence which was not confirmed histologically. In June I967 the transverse colostomy was closed and the patient was discharged from the hospital in July x967. He was seen in January I969 apparently free of carcinoma and well up to the present time (Fig. I, B).
380
BRITISH JOURNAL OF PLASTIC SURGERY
A
B
FI~. I Case I . - - A , Large elevated tumour masses and sinuses of the skin on the dorsal aspect of the left buttock. B, Two years after radical surgery and primary skin grafting.
A
B
FIG. 2 Case 2.--A~ Irregular-shaped fistulae and turnout masses in the right buttock. Be Completely healed upper part of the buttock after skin grafting. In the lower part there is recurrence of the tumour.
SQUAMOUS CELL CARCINOMA IN PERIANAL PYODERMA
381
Case 2.--A 59-year-old man, a truck driver, had acne conglobata involving the buttocks for 3° years. In June 1967 he was seen in the Department of Plastic Surgery complaining of severe pain in the right gluteal region in which area he had observed tumour changes. Physical examination revealed many irregular fistula: with tumour masses in the right gluteal area. Blood and pus were oozing over the infected right buttock. Biopsy showed epidermoid squamous cell carcinoma (Fig. 2, A). In August 1967 an extensive resection of the skin, subcutaneous tissue and gluteal muscles of the right buttock including the tumour masses was performed. The defect was covered with a thin split thickness skin graft taken from the thigh. The take of the graft was 8o per cent. A regrafting procedure was performed three weeks later. The patient was discharged to the Department of Dermatology for follow-up. In the right groin small lymph nodes were palpable. In September 1967 radiotherapy was given for metastases in the nodes of the right groin. A total of 5746 r or 281-291 r/dose was given. The grafted area was completely intact. In January 1968 an operation for local recurrence and a radical groin dissection was planned. However, the patient died on the operating table in cardiac arrest. Resuscitation and heart massage (3 hours) were unsuccessful. An autopsy was performed but no sign of tumour in other organs was seen (Fig. 2, B). C o m m e n t s . - - E p i d e r m o i d cancer may occur (I) in untreated anal fistula: as well as following fistulotomy, (2) in hidradenitis suppurativa and (3) in certain venereal diseases (Turell, 1962). In both cases described a period of 27 years and 30 years of chronic inflammation in the perianal region occurred. During this time the inflammation resisted all kinds of conservative treatment and fistulotomy. T h e malignant change seemed to occur in both cases in the fistula: of the sinuses themselves. The carcinomata were slow to grow and metastasise. It seems justifiable to plead for a more radical operative approach to chronic fistula: occurring in ache conglobata as well as in chronic anal fistula:. Wide excision of the affected area is advocated (Turell, 1962 ; Dillon and Spjut, 1964). T h e defect may be left open, but we prefer to cover it immediately with split thickness skin grafts which shortens the time in hospital. SUMMARY Two cases of epidermoid squamous cell carcinomata arising in chronic perianal pyoderma are described. Radical surgery would appear advisable in patients with relapsing draining sinuses and anorectal fistula: rather than unroofing procedures. T h e authors used free skin grafting on the tissue defect in order to achieve rapid wound healing. REFERENCES DILLON, J. S. and SPJUT, H. J. (I964). Ann. Surg. I59, 45I. HELLIER~ F. A. (I939)- Br. J. Derm. 5I, Io9. JACKMAN,R. J. and McQUARRIE, H. B. (I949). Am. J. Surg. 77, 349. M6LLER, C., SAKSELA,E. and SUNDELL,B. (I969). Acta chit. scand. I35 , 83. ROSSER, C. (I93I). A m . J . Surg. I I , 328. RIJNDLE, F. F. and HALES, I. B. (x953). Ann. Surg. I37, 2I 5, SCHIFF, B. and KERN, A. B. (I957)- Archs Derm. 75, 878. SGHWARZ,A. (1963). Dis. Colon Rectum, 4, 305. TURELL, R. (I962). Surg. Clins N. Am. 42, I235.
Department of Plastic Surgery, First Surgical Clinic, University Hospital, Helsinki, Finland SQUAMOVS cell carcinoma in chronic perianal pyoderma is a rare lesion. There are only a few reports on carcinoma arising secondary to ache conglobata (Hellier, I939 ; Schiff and Kern, I957; Dillon and Spjut, I964) and to anorectal fistulae (Rosser, i 9 3 i ; Rundle and Hales, x953 ; Schwarz, I963). Chronic inflammation in the perianal skin and subcutaneous tissue may continue for a long period without affecting the general health or the working capacity. Similar clinical features (M611er, Saksela and Sundell, I969) can be produced by ache conglobata, hidradenitis suppurativa, anal fistula: and pilonidal sinuses and the correct origin may be difficult to determine because of the inflammation (Jackman and McQuarrie, 1949). Carcinoma arising in an area of untreated or unhealed fistula: and draining abscesses permits early diagnosis by inspection and palpation. I f any area of hard induration is noted, especially if combined with ulceration of the skin, the suspicion of malignant change should be aroused and a biopsy done (Turell, I962). This is a report of two cases with uniform clinical appearance but with different histological background which developed squamous cell carcinoma after years of chronic inflammation. Case I.--A 52-year-old serviceman was first seen by us in February I967. Perianal pyoderma draining through his gluteal region developed in x94o. The fistulous tract was unroofed in I94I but never healed completely. In I964 multiple abscesses developed with sinuses draining a foul-smelling mixture of pus and blood over the gluteal region. He was seen in another hospital for conservative treatment at that time. In December I966 a ha~morrhage from the left buttock brought the patient to the II Surgical Clinic, University Hospital, Helsinki. The whole left gluteal region was infected and indurated (Fig. I, n). There were large, elevated fungating tumour masses on the upper part of the left buttock. On the medial side~ a large anorectal fistula was observed. Biopsy of the tumour showed a keratinous type of epidermoid carcinoma. Fismlography revealed a fistula immediately above the anal sphincter, directed towards the left buttock. On sigmoidoscopy, inflammation of the rectal mucosa was seen but no malignant disease could be observed. In February x967 a transverse colostomy was made ; the patient was then admitted to the Department of Plastic Surgery for further treatment of his left buttock. In March I967 a wide resection of skin, subcutaneous and muscular tissue including the tumour masses and the sinuses of the skin of the left buttock was performed. The whole tumour area was removed up to the anal sphincter which was left intact. The tissue defect extended over the whole left buttock and an area around the anal canal. The defect was immediately covered with a rotation flap from the lateral aspect of the buttock and partly with a medium split thickness skin graft from the thigh. The flap and the graft healed well. InApril x967 an abdomino-perineal resection was performed. Apermanent sigmoidostomy was left. No metastases could be found at that time, Carcinoma of the colon could not be verified ; only fibrosis and granulation tissue appeared in the histological specimens. In May I967 the patient was readmitted for suspected recurrence which was not confirmed histologically. In June I967 the transverse colostomy was closed and the patient was discharged from the hospital in July x967. He was seen in January I969 apparently free of carcinoma and well up to the present time (Fig. I, B).
380
BRITISH JOURNAL OF PLASTIC SURGERY
A
B
FI~. I Case I . - - A , Large elevated tumour masses and sinuses of the skin on the dorsal aspect of the left buttock. B, Two years after radical surgery and primary skin grafting.
A
B
FIG. 2 Case 2.--A~ Irregular-shaped fistulae and turnout masses in the right buttock. Be Completely healed upper part of the buttock after skin grafting. In the lower part there is recurrence of the tumour.
SQUAMOUS CELL CARCINOMA IN PERIANAL PYODERMA
381
Case 2.--A 59-year-old man, a truck driver, had acne conglobata involving the buttocks for 3° years. In June 1967 he was seen in the Department of Plastic Surgery complaining of severe pain in the right gluteal region in which area he had observed tumour changes. Physical examination revealed many irregular fistula: with tumour masses in the right gluteal area. Blood and pus were oozing over the infected right buttock. Biopsy showed epidermoid squamous cell carcinoma (Fig. 2, A). In August 1967 an extensive resection of the skin, subcutaneous tissue and gluteal muscles of the right buttock including the tumour masses was performed. The defect was covered with a thin split thickness skin graft taken from the thigh. The take of the graft was 8o per cent. A regrafting procedure was performed three weeks later. The patient was discharged to the Department of Dermatology for follow-up. In the right groin small lymph nodes were palpable. In September 1967 radiotherapy was given for metastases in the nodes of the right groin. A total of 5746 r or 281-291 r/dose was given. The grafted area was completely intact. In January 1968 an operation for local recurrence and a radical groin dissection was planned. However, the patient died on the operating table in cardiac arrest. Resuscitation and heart massage (3 hours) were unsuccessful. An autopsy was performed but no sign of tumour in other organs was seen (Fig. 2, B). C o m m e n t s . - - E p i d e r m o i d cancer may occur (I) in untreated anal fistula: as well as following fistulotomy, (2) in hidradenitis suppurativa and (3) in certain venereal diseases (Turell, 1962). In both cases described a period of 27 years and 30 years of chronic inflammation in the perianal region occurred. During this time the inflammation resisted all kinds of conservative treatment and fistulotomy. T h e malignant change seemed to occur in both cases in the fistula: of the sinuses themselves. The carcinomata were slow to grow and metastasise. It seems justifiable to plead for a more radical operative approach to chronic fistula: occurring in ache conglobata as well as in chronic anal fistula:. Wide excision of the affected area is advocated (Turell, 1962 ; Dillon and Spjut, 1964). T h e defect may be left open, but we prefer to cover it immediately with split thickness skin grafts which shortens the time in hospital. SUMMARY Two cases of epidermoid squamous cell carcinomata arising in chronic perianal pyoderma are described. Radical surgery would appear advisable in patients with relapsing draining sinuses and anorectal fistula: rather than unroofing procedures. T h e authors used free skin grafting on the tissue defect in order to achieve rapid wound healing. REFERENCES DILLON, J. S. and SPJUT, H. J. (I964). Ann. Surg. I59, 45I. HELLIER~ F. A. (I939)- Br. J. Derm. 5I, Io9. JACKMAN,R. J. and McQUARRIE, H. B. (I949). Am. J. Surg. 77, 349. M6LLER, C., SAKSELA,E. and SUNDELL,B. (I969). Acta chit. scand. I35 , 83. ROSSER, C. (I93I). A m . J . Surg. I I , 328. RIJNDLE, F. F. and HALES, I. B. (x953). Ann. Surg. I37, 2I 5, SCHIFF, B. and KERN, A. B. (I957)- Archs Derm. 75, 878. SGHWARZ,A. (1963). Dis. Colon Rectum, 4, 305. TURELL, R. (I962). Surg. Clins N. Am. 42, I235.