Review

ORAL FLORID PAPILLOMATOSIS (VERRUCOUS CARCINOMA) DAVID GRINSPAN, M.D. AND |ORGE ABULAFIA, M.D.

From the Department of Dermatology Rawson Hospital and Durand Hospital Buenos Aires. Argentina

In 1960, Rock and Eisher' and subsequently Wechsler and Fisher^ suggested tbe term oral florid papillomatosis (EP) for a process characterized by lesions in the mouth and larynx. These lesions were either single or multiple, and in the latter case appeared either simultaneously or successively, accompanied by peculiar clinical, histologic and evolutionary characteristics. Clinically, they were vegetating and sometimes verrucous lesions that simulated carcinomas. Histologically, they presented acanthosis and papillomatosis, while in the inferior layers of tbe epithelium, some lesions showed poikilocytosis, poikilokarynosis and numerous typical mitosis. However, there were neither loose strands nor rupture of the basal layer that might suggest an infiltrating carcinoma. These lesions showed an invasive tendency, as seen in true carcinomas, and there were instances of bone invasion and fistulization of tbe skin. Nevertheless, and this was a highly important sign, no metastatic spread was recorded. Surgery was considered the treatment ot choice, although recurrences were frequent, rendering necessary additional operations. Failures were recorded with podophyllum. Address for reprinis: David Grinspan, M.D., Department of Dermatology, Rawson Hospital, Buenos Aires, Argentina.

electrocoagulation and autovaccines with tumoral tissue. Investigators*-^ compared EP with a laryngeal papilloma of the adult, that may undergo malignant transformation after radiotherapy. The condition was also compared with papillomas of the penis and uterus. EP was differentiated from an in situ epithelioma that presents atypical cells throughout the epithelium, and from verrocous leukoplakia, since EP was not regarded as a tumor of keratotic nature.'-^ The electron microscope revealed differences between EP and a classical carcinoma. No viruses were found by Rock and Eisber. In 1963, Samitz and Weinberg^" reported 80% improvement of a case of EP of the buccal mucosa following the administration of 0.5 mg aminopterin daily for 7-10 days, alternating with 7-10 days of rest. This patient was treated for 22 months, but eventually developed a carcinoma.'"' In Argentina, Jaimovich et al.^ reported tbe sixth case of EP in the world literature, localized in the mucosa of both cheeks with fistulae extending to the surface. Subsequently, Kaminsky et al.^ reported successful results with methotrexate in a patient witb a lesion with atypical mitoses. Current Cases

We have observed 32 cases of EP over a two year period.^^ While gathering data, we could not help but wonder about this high incidence when compared with relatively few cases published so far. We raised the question whether

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EP was a new pathologic entity, or whether it had been wrongly diagnosed as differentiated squamous cell carcinoma or verrucous leukoplakia. In reviewing the world literature, we found cases reported by Ackerman**"'' presenting lesions identical to EP and described as verrucous carcinoma. These authors studied 31 cases (25 men and six women) with an average age of 67 years. Eighteen of these patients chewed or smoked tobacco; seven had leukoplakia, while 50% evidenced poor dental hygiene. There were subsequent publications on verrucous carcinoma, until 1966, when Kraus and Perez-Mesa" reported 105 cases of which 77 were localized in the oral mucosa. The remainder were noted in the larynx, penis, nostrils, vulva and vagina, scrotum and perineum. Eight of the cases underwent carcinomatous transformation (four after radiotherapy). Thus, it appears that EP closely resembles the already known verrucous carcinoma** which in our opinion is a low-grade squamous cell carcinoma with a rare tendency for metastasis. EPmay also resemble otber lesions: (1) the type II Buschke Eowenstein's giant condyloma of the genitalia"' that at times manifests as a true condyloma accuminatum and at other times as a squamous cell carcinoma with a very special behavior; (2) GrissonDelbanco's" condylomatoid precancerosis; (3)Civatte's horny papillary epithelioma'^; (4) Ewing's squamous cell diffuse papillomatosis'^; (5) Gottron's papillomatosis cutis carcinoides'Mhat was considered by different authors as a secondary carcinoma developing in a scar (Marjolin's ulcer, lupus lesions, etc.). Vilanova et al.'"' believed that EP resembled AzOa's pseudo-epithelloma that is in reality a vegetating pyodermitis; (6) Some cases of chronic hyperplastic candidiasis with multiple foci'5-'^; (7) Confluent or multiple squamous cell papilloma of [he larynx and respiratory tract; (8) diverse processes described under different denominations, such

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as: (a) verruca accuminata of the tongue and squamous cell carcinoma^"; (b) oral papillomatosis^'-^''; (c) Schneider's papilloma of the nostrils"; Schneicher-Gottron's papillomatosis mucosa carcinoides^^; and Ronchese's leukoplakia-leukokeratosis.^** The term EP was also used to designate certain nodular lesions of the nipple attributed to papillomatosis of the milk ducts." This disease in not related to EP. An example of the "rediscovery" of verrucous carcinoma and its further notoriety under the denomination of EP is exemplified by one of our patients. He presented an enormous destructive tumor in the buccal mucosa that extended to the upper jaw and to the skin. This tumor was accompanied by metastatic lymph nodes and was interpreted as a carcinoma of very difficult management. A surgical approach would have proved very destructive, without any assurance of a definite cure; on the other hand, radiotherapy would have been merely palliative. Inasmuch as the histologic study corresponded partially to EP, we started treatment with methotrexate and the lesion was reduced to ab