Buschke-Loewenstein (Giant Condylomata WILLIAM DREYFUSS,
M.D.
AND
WILLIAM E. NEVILLE,
1896 Buschke’ described a form of condyIomata acuminata with the cIinica1, but not the histoIogic appearance of peniIe carcinoma. Whereas Buschke was the first one to anaIyze the condition cIearIy, he was not the first one to observe and report it. In 1804 Martens and TiIesius12 published an AtIas of VenereaI Diseases. They show a picture (Fig. I 7 of their articIe) which reminds one of the disease with which we are concerned. Ricordn in 1851 wrote a treatise on venerea1 diseases. In Figure 48 of his articIe he pictures what he describes in his own words as “vegetations frambesiees avec phimosis et perforation du (The Iatter two sources were not prepuce.” avaiIabIe in original form and are quoted from Frei.) Ricord cautioned not to confuse these Iesions with carcinoma. Considering the status of the diagnostic means at his command this was indeed a remarkabIe observation. For the same reason we shouId not hoId it against the authors if they incIude the disease among those caIIed “venereal.” : In Iater years Buschke and his co-worker Loewenstein as we11 as Frei, MuehIpfordt and Treite wrote of this medica phenomenon.2-4s13,14,16 In this country Gersh6s7 devoted severa pubIications to the same probIem. CIinicaIIy, one is deaIing with a Iesion of the folIowing characteristics: inside a phimotic prepuce deveIops what Iooks Iike an ordinary condyIoma acuminatum. Soon nodular areas of induration appear in the prepuce and Iater in the skin of the peniIe shaft, particuIarIy in its dorsa1 aspect. Gradually perforations occur in the periphery of these noduIes. Out of the resuIting IistuIas sprout new condyIomas and putrid smeIIing pus. The fistuIas merge and leave Iarge hoIes in the skin of the penis and prepuce. EventuaIIy the penis may more or Iess be grown over with condyIomatous masses. In an ordinary condyIoma the strata are histoIogicaIIy we11 differentiated and the epitheIia1 prohferation never goes beyond a cer-
I
N
Tumors Acuminata) M.D.,
Cleveland,
Ohio
tain depth. This is basicaIIy the structure of a so-caIIed giant condyIoma. The onIy important histoIogic as we11 as cIinica1 distinction is the tendency to grow into deeper tissue Iayers. This growth in depth is, however, strictIy one by expansion and not infiItration. NevertheIess, the tissue in front of the on-coming condyIoma masses, particuIarIy the corpora cavernosa, can be destroyed by compression. There is aIways an area of “skirmishing” round ceIIs to be found between the norma tissue and the condyIoma. AIthough one may find a few epitheIia1 pearIs, there is no disorderIy structure or growth, no invasion of Iymph or bIood vesseIs and no metastasis. Any Iymph node enIargements are due to inffammation. CertainIy the destruction of tissue is a sign of maIignancy but it is of utmost importance whether it is done by expansion or carcinomatous infiItration. This problem has caused some confusion in the Iiterature. The point in question cIearIy is whether or not these Iesions are precancerous. FreudenthaI and Spitzer5 taIk about “condyIomatoid precanceroses” and “condyIoma-1ike carcinoma.” MuehIpfordt13 aIso uses the same He argues that the histoIogic expression. diagnosis shouId not be decisive and prefers what he caIIs the “much simpIer and safer clinica conception of maIignancy.” He cites two cases, one of IsraeI’O in which a truIy precancerous condition existed. However, this cIearIy showed up in the microscopic sIide. In the second case, the first biopsy showed condyIoma and the second proved to be carcinoma of the frenuIum. It is a basic ruIe that biopsies have to be extensive and there is always a possibiIity of the described carcinoma of the frenuIum having been the primary Iesion, causing the formation of secondary condyIomas by its decomposition. The same can be said about the case reported by Herman.g No one can contest the fact that any fibroepitheIioma, as a condyloma is, can occasionaIIy
Buschke-Loewenstein
without sequelae. He was never circumcised and was perfectIy we11 unti1 about mid-October, 1950. He then observed that his penis began to sweI1 and became hard around the edges of the prepuce. He was unabIe to puI1 his foreskin back and a sIight discharge appeared. Soon he feIt an indurated area on the dorsal portion of the shaft of the penis about in mid-length. He was treated with penicillin injections. On December 13, 1950 he was hospitalized because there was no response to treatment. Physical examination was negative except for focal findings. BIood pressure was 108/62. Temperature ranged between 98” and 99%. during the whoIe period of hospitalization. Urinalysis revealed the folIowing: pH 4.5, aIbumin negative, sugar negative. Microscopic examination showed a few white and red bIood cells. KIine test was doubtfu1 (repeat was negative). Sedimentation rate was 60 mm. in 60 minutes. HemogIobin was 13.5 gm. per cent. White blood count was 10,500 with eosinophiIs 3 per cent, stab forms 2 per cent, segmenta neutrophiIs 75 per cent, Iymphocytes 13 per cent and monocytes 3 per cent. Th e penrs’ was swoIIen to three times its norma size and there was considerabIe edema. The urethral orifice was just visible in the ring of the much enIarged prepuce. The middle third of the penile shaft was very indurated but not tender and there were four areas of ulceration with a central depression and a periphera1 corona of cauliflower type granulations, which readily bIed. The largest one of these areas measured 2.5 by 2 cm. A foulsmelling white-yellow pus oozed from the prepuce and the perforations. The remaining skin of the shaft was very tight and there \T’as bilateral, painless, inguina1 adenopathy. On December I 3, 1950, a dorsal slit was done. The prepuce was found to be filled with condylomas. Biopsies were taken from the prepuce and the shaft tissue. Pathologic report indicated heavily inflamed Ioose connective tissue lined on one side by masses of papilIary structures, with marked hyperpIasia and hypertrophy of the MaIpighian layers of covering squamous ceI1 epitheIium with acanthosis, mild parakeratosis and balIoonization of cells. There were numerous bands of deIicate connective tissue within the narrow stalks of papiIIae extending between the epitheIia1 masses into the surface. In the stalks were round and mononucIear cells, sometimes perivascularIy
progress into a carcinoma. It is possibIe that in MuehIpfordt’s case such a condition existed. He taIks of Unna-Delbanco disease and actually describes an acanthoma which is something different than a condyIoma acuminatum. The term “precancerous” cannot be used loosely. According to Borst and Meyer one would have to conceive that transformation of a certain tissue into carcinoma is the rule and not the exception. Treite had observed condyIomata acuminata with severa recurrences over a period of tweIve years. FinaIIy squamous cell carcinoma deveIoped. Quoting the aforementioned thesis of Borst and Meyer,. he stiI1 declined to caI1 the primary lesion precahcerous. EtioIogicaIIy no clear-cut expIanation for the formation of condyIomas has been advanced. There is the possibility of a fiItrable virus. However, there is aIso no doubt that the moisture and irritation of the smegma in the preputia1 space is at Ieast a strong supporting factor as Gersh has stressed. TREATMENT
Radiation treatment has been tried without success. One may hesitate to use podophyIlin in such an extensive lesion. Fulguration has been used and evidentIy was sufficient in some cases. Surgery is the treatment of choice. It may vary from IocaI excision by knife or eIectroIoop to amputation of the penis. A comparatively large number of amputations of the penis are cited in the Iiterature. Some cases are reported cured with Iess formidabIe methods. Frei, who reported five cases treated by fulguration, desiccation and circumcision, is the onIy author mentioning a plastic repair using scrotal skin. No argument shal1 be presented against amputation because it is extremely diff&It to judge a case without having actuaIIy observed it. The large number of recurrences make it mandatory to remove all pathologic tissue in the first stage of any treatment. We present our case because its outcome shouId encourage an attempt of radica1, yet conservative treatment even in an advanced stage of the disease. CASE
Tumors
REPORT
W. D. was a thirty year oId, white, unmarried man. His famiIy and persona1 history were non-contributory. Fifteen years prior to admission he had gonorrhea and was cured I47
Dreyfuss
and NeviIIe
FIG. I. Photograph showing the condytomas of the preputial sac and the dorsa1 shaft perforation; the other ones being IateraI are not visible. (P illustrates the
FIG. 2. Inflammatory
reaction
1940. Treatment with antibiotics IocaIIy and by injection was continued. The pathoIogic process progressed and he was seen by us on February I 5, 1951. The description of the IocaI Iesion wouId be the same as the one on his first hospita1 admission, onIy that the areas of ulceration had enIarged and foIlowing the dorsa1 slit the glans was more visibIe. (Fig. I.) CIinicaI impression was peniIe carcinoma. The patient was hospitaIized on February 19, 194’. Genera1 examination reveaIed nothing new. In genera1 the Iaboratory findings were simiIar to the ones at first admission. On February zI.st an extensive biopsy in depth was made; hemorrhage had to be controIIed by fuIguration. The specimen, 2 by 1.3 by 0.7 cm., was again diagnosed as “condyIoma acuminatum.” The first pathoIogist submitted it to another institution. The diagnosis was the same. In spite of the wide extension we decided to try preservation of the penis but were convinced that we shouId sacrifice the Iarger portion of the skin and do a secondary pIastic procedure. On February 24th the operation was performed under genera1 anesthesia. After preparation and draping, a tourniquet was pIaced on the base of the penis. The skin was excised about 135 cm. distance from the base to the gIans. This incIuded a11 the fungating masses. LateraIIy an incision was made at
at base of condyloma.
In the grooves between the papiIIae much parakeratotic, sometime hyaIinized matter and bIood was found. In the subepithelia1 spaces there was considerabIe round and mononucIear ceI1 infltration 0ccasionaIIy in perivascuIar arrangement, aIs enIargement of vascuIar spaces. There was no invasion of epitheIium into the underIying tissue. Occasionally, smaI1 granuIoma-Iike structures were seen in the subepitheIia1 connective tissue, consisting mainIy of round, mononucIear and, rareIy, giant ceIIs of foreign body type. Diagnosis was condyIoma acuminatum of foreskin of penis with extensive papiIIary formations and acanthosis, occasionaIIy perivascuIar arrangement of ceIIs. The patient was discharged on December 22,
arranged.
148
Buschke-Loewenstein
Tumors
FIG. 3. Stage I of pIastic repair with bridge flap.
FIG. 4. Final result.
least !i cm. into normal tissue. This left a narrow bridge of cutaneous covering over the corpus spongiosum. The fascial layer was removed and the corpora cavernosa were exposed. On one area, dorsaIIy to the left of the midline, the condyIomatous tissue had expanded into the corpus cavernosum. There we had to open and remove part of the corpus in order to be radica1. Anteriorly the corona was sacrificed and on one spot a smaI1 portion of the gIans, which otherwise was remarkabIy free of disease. AI1 together 85 gm. of tissue was removed, the Iargest condyIomatous area measuring 7 by 4 by I cm. (PathoIogic diagnosis again was condyIoma acuminatum.) (Fig. 2.) The edges of the gIans were fuIgurated. The defect in the Ieft corpus cavernosum was cIosed with Iockstitch suture and the tourniquet removed. The Iarger vessels were cIamped and tied. Other bIeeding points were taken care of by suture Iigation and fulguration. The skin on the Iower surface as we11 as on the base was prevented from retracting by a few situating sutures. The wound was treated with aureomycin ointment and the patient was pIaced on oral medication with the same drug, 230 mg. every six hours for ten days. He was discharged on February 28, 194 I. After five weeks, the wound was cIean, a11 catgut having been dissolved or absorbed, and there were heaIthy granuIations with no signs of recurrence. for On ApriI 3, 194 I, he was hospitaIized stage I of the pIastic repair operation which was performed on ApriI 4th under genera1 anesthesia. After preparation and draping, redundant granuIations were removed from the peniIe wound. The skin edges on the dorsa1 portions of the gIans and the base were fresh-
ened. A bridge flap about 3 cm. in width was formed from the anterior aspect of the left side of the scrotum, Ieaving both ends attached to the scrota1 skin, but with sufficient cIearance to accept the penis comfortabIy. The edges were attached to the gIans and skin of the peniIe base. (Fig. 3.) Again aureomycin dressings were appIied and ora medication with the same drug was given. The patient was discharged on ApriI 5, 1951. The ffap took weI1. In the subsequent admissions, first the left piIIar of the bridge and Iater the right one was severed after test and the edges united with the smaI1 skin island on the lower surface of the penis, the gIans as we11 as the skin of the peniIe base. 1951, the transpIant began to By August, thin out and the penis appeared fairIy normal. InguinaI Iymphadenopathy had subsided. (Fig. 4.) FunctionaIIy there has been no complaint. The patient was married on December 25, 195 I, He has been seen repeatedIy since and to date there is no sign of recurrence.
COMMENTS
So-caIIed giant condyIomata acuminata “ Buschke-Loewenstein tumors” are a rare variation of simpIe condyIomata acuminata and, Iike these, generaIIy originate in the preputial sac. HistoIogicaIIy they appear alike except for a pronounced front of round ceI1 infiItration between the papiIIomatous structure and the norma tissue. Whether these round ceIIs themseIves pIay a roIe in the rapid expansion of these tumors or whether they are the expression of a yet unknown virus type I49
Dreyfuss agent remains unknown. The tumors are easiIy mistaken for peniIe carcinoma because of their rapid growth and tendency to involve outside of the prepuce the whoIe shaft of the penis. A pure cIinica1 conception of the disease has Ied to a certain confusion in nomenclature. The terms “giant condyIomas” or “carcinomaIike condylomas” are perfectIy in order. However, one must absoIuteIy reject the cIassification of “condylomatoid precancerosis.” It must be kept in mind that the aggressiveness of these tumors is by expansion and penetration, not by infiItration as in the case of a carcinoma. The histoIogic diagnosis of proper biopsies must guide our pIan of treatment. Because their growth is so rapid, we advocate earIy, radica1 yet preserving surgery, meaning remova of a11 involved skin into norma tissue (in width as we11 as in depth) even if it shouId invoIve partial resection of the corpora cavernosa. PIastic procedures can foIIow as soon as the disease is controIIed. SUMMARY
A case of giant condyIomata acuminata with extensive destruction of penile skin and expansion into one corpus cavernosum is presented. Radical remova of a11 diseased tissue into norma structures and secondary pIastic procedure resulted in cure. PathoIogy, nomencIature and treatment are discussed. REFERENCES I. BUSCHKE, A.
Neisser’s Stereoskopischer AtIas, 1896. Cited by Buschke and Loewenstein.2 2. BUSCHKE, A. and LOEWENSTEIN, L. Uber car-
and NeviIIe cinomlhnliche Condylomata Acuminata des Penis. Klin. Wcbnscbr., 4: 1726, 1925. 2. BUSCHKE. A. land LOEWENSTEIN. L. Uber die Beziehingen bon spitzen KondyIbmen zu Karzinomen des Penis. Deutscbe med. Wcbnscbr., 58:
809, ‘932.
4. FREI, W. Uber “Carcinoma%_nIiche” spitze CondyIome am Penis. Arch. j. Dermat. IL. Sypb., 160: ‘09,
‘930.
5. FREUDENTHAL, W. and SPITZER, R. Warzen und KondyIome. Handb. d. Haut-u. Gescblecbtskr., 12: 33, ‘933. 6. GERSH, I. CondvIomata acuminata of the maIe exteinal genitaba: an effective method of surgical treatment. Ural. @ Cutan. Rev., 49: 432, 1945. 7. GERSH, I. Giant condylomata acuminata (carcinema-Iike condylomata or Buschke-Loewenstein tumors of the penis). J. Ural., 69: 164, 1953. 8. GRISSON and DELBANCO, E. Monstriiser Tumor der Genitalgegend. Dermat. Wcbnschr., 60: 89, 1915. o. HERMAN. L. The Practice of Uroloav. PhiIadeIohia. 1945. W. B. Sanders Company. IO. ISRAEL, W. Zur Kenntnis der atypischen CondyIomata Acuminata des Penis. Ztscbr. j. Ural., 221: 395, 1928. I I. LOEWENSTEIN, L. Carcinoma-like condyIomata acuminata of the penis. M. Clin. North America, I
Y”
a
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23: 789, 1939. 12. MARTENS and TILESIUS. Atlas d. ven. Krankheiten, 1804. Cited by Frei.4 ‘3. MUEHLPFORDT, H. Uber die KondyIomatoide der Genitalgegend (DeIbancoPrgkanzerose Unna). Dermat. Wcbnscbr., 87: 1403, 1928. und KondvIome” IL MUEHLPFORDT. H. “Soitze Karzinom iA selben Tumor. K&k, HistoIogie und Therapie der kondylomatoiden Prgkanzerose dcr GenitaIgegend (DeIbanco-Unna). Dermat. Wcbnscbr., 93: I 145, 1931. IS. , RICORD and TRAIT& ComoIet des maIadies v&n&riennes, 185 I. Cited by Frei.4 16. TREITE, P. iiber die Karzinomentstehung auf spitzen Kondylomen. Zentralbl. j. Gyniik., 65: 1096, Ig4I. 17. UNNA, P. G. Die HistopathoIogic der Hautkrankheiten. Berlin, 1894.