SOLITARY BASAL CELL NEVUS STAGE OF BASAL CELL EPITHELIOMA

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1 SKIN RESEARCH Dec Vol.10 No SOLITARY BASAL CELL NEVUS PREINVASIVE STAGE OF BASAL CELL EPITHELIOMA YOSHIHARU MIKI, M.D. Department of Dermatology, University of Osaka, School of Medicine, Osaka, Japan (Director: Prof. T. FUJINAMI) INTRODUCTION Basal cell epithelioma has been reported to develop from chronic radiodermatitis, xeroderma pigmentosum, or the nevus sebaceus of JADASSOHN.1) In addition to these precancerous dermatoses, GORLIN's or basal cell nevus syndrome, which consists of multiple basal cell nevi of the skin, skeletal anomalies, and intracranial lesions,2) develops multiple, invasive basal cell epithelioma later. In this sense, GORLIN's syndrome should also be included among the precancerous dermatoses for the basal cell epithelioma. SUMMERLEY,3) in a series of 125 basal cell epithelioma cases, aged 45 or under, 110 cases of which were solitary lesions, found dental jaw cysts, cervical ribs, or other skeletal anomalies in 7 cases, suggesting of a close relationship between the solitary basal cell epithelioma and the forme fruste of GORLIN's syndrome. In the present report, attempts will be made to disclose a preinvasive stage of some of the longstanding, solitary basal cell epithelioma. The preinvasive stage is considered to be separable from the invasive stage and to be designated as a solitary basal cell nevus. MATERIALS AND METHODS Among the 63 cases of basal cell epithelioma histologically diagnosed between 1956 and 1966 at the Department of Dermatology of the University of Osaka, 3 cases were associated with xeroderma pigmentosum and 6 cases with chronic radiodermatitis. The remaining 54 cases, 52 cases of which were solitary lesions, were found in apparently normal skin with no obvious precancerous conditions. In 8 of the 54 cases, the patients stated that the lesions were of many years' duration. Earlier snap-shots or memorial photographs of known dates were submitted from these 8 cases to compare the previous with the present status of the tumors. Fortunately, most of the tumors were grossly pigmented and could easily be identified on black and white pictures.

RESULTS CASE II CASE I A 50-year-old school teacher with a 1.2 ~ 0.8 cm pigmented basal cell epithelioma on the right upper lip stated that the lesion had been present longer than he could remember.

2 RESULTS CASE II CASE I A 50-year-old school teacher with a 1.2 ~ 0.8 cm pigmented basal cell epithelioma on the right upper lip stated that the lesion had been present longer than he could remember. Pictures taken at 45, 46 and 47 years of age showed a smaller pigmented lesion. There was a rapid growth of the lesion after 47 years of age (Fig. 1). No such lesion was visible in a picture taken at 44 years of age. A 26-year-old man with a 1.0 ~ 0.7 cm pigmented basal cell epithelioma at the upper border of a round depressed scar on the right cheek stated that the lesion had been present for more than 6 years. The picture taken at 19 years of age showed no such lesion, but the one taken 10 months later showed a small pigmented lesion. Successively taken pictures at 22 and 25 years of age showed that the lesion had become enlarged, then was irradiated but recurred (Fig. 2). FIGURE 2 (Case II) Basal cell epithelioma at 26 years of age (5). Note a preceding lesion at 19 10/12 (2), 22 (3) and 24 (4) years of age. No such lesion is visible at 19 0/12 years of age (1). FIGURE 1 (Case I) Basal cell epithelioma at 50 years of age (4). Note a smaller preceding lesion in the right upper lip at 45 (1), 46 (2) and 47 (3) years of age.

CASE III A 44-year-old department-store manager stated that a pigmented mole had been present on the left cheek since childhood. The lesion was a 1.0 ~ 0.

3 CASE III A 44-year-old department-store manager stated that a pigmented mole had been present on the left cheek since childhood. The lesion was a 1.0 ~ 0.7 cm pigmented and semitransparent nodule and there had been a rapid enlargement during the previous several months. In a snap-shot taken at 35 years of age, a smaller pigmented lesion was identified (Fig. 3). There were no earlier pictures available. Histologically, the lesion was a well circumscribed mass of interwining nests and strands of basal cell epithelioma with abundant stroma. No inflammations or ulcerations were present (Figs. 4 & 5). FIGURE 4 Hematoxylin-eosin stained section of basal cell epithelioma in Case III. 565F64, ~ 9. FIGURE 5 Higher magnification of Fig. 4. Hematoxylin-eosin, ~ 68. FIGURE 3 (Case III) Basal cell epithelioma at 44 years of age (2). Note a smaller preceding lesion at 35 years of age (1).

4 CASE IV A 61-year-old chief-guard at the School of Medicine had a 0.5 ~ 0.4 cm ulcerated and pigmented basal cell epithelioma on the right ala nasi. He stated that the lesion had been present as long as he could remember. However, no such pigmented lesion was visible in the picture taken at 28 years of age. A definitely black lesion was identified in a snap-shot taken at 45 years of age (Fig. 6). CASE V A 65-year-old man stated that he had had a black mole on the bridge of his nose since he was a child. The lesion became ulcerated during the previous several years and measured 2.3 ~ 1.6 cm at the time of examination. A snap-shot taken at 62 years of age showed a smaller pigmented lesion in the same area (Fig. 7). CASE VI A 47-year-old latheman stated that he had had a black mole near the hair line of the left temple since childhood. The mole had become FIGURE 6 (Case IV) Basal cell epithelioma at 61 years of age (3). Note a smaller preceding lesion at 45 years of age (2). No such lesion is visible at 28 years of age (1). ulcerated several years prior to examination and had been electrodesiccated by a local physician. The lesion recurred and by the time of examination it had become a 2.0 ~ 2.0 cm fungating tumor with an eroded surface and a pigmented rolled border. A snap-shot taken at 40 years of age clearly showed a smaller pigmented lesion (Fig. 8). CASE VII A 71-year-old man had a mole on the left cheek since childhood. The mole had become ulcerated and had been excised about 4 years prior to examination but soon recurred. The lesion measured 4 ~ 5 cm and was a fungating eroded tumor with a rolled pigmented border FIGURE 7 (Case V) Basal cell epithelioma at 65 years of ge (2). Note a smaller pigmented lesion at 62 years of age (1).

FIGURE 8 (Case VI) Basal cell epithelioma at 47 years of age (2). Note a smaller preceding lesion at 40 years of age (1). FIGURE 9 (Case VII) Basal cell epithelioma at 71 years of age (3).

5 FIGURE 8 (Case VI) Basal cell epithelioma at 47 years of age (2). Note a smaller preceding lesion at 40 years of age (1). FIGURE 9 (Case VII) Basal cell epithelioma at 71 years of age (3). Note a smaller pigmented lesion at 51 (1) and 66 (2) years of age. FIGURE 10 (Case VIII) Basal cell epithelioma at 71 years of age (3). Note a smaller preceding lesion at 51(1) and 68 (2) years of age. 683

6 at the time of the examination. Both photographs taken at 51 and 66 years of age showed the lesion (Fig. 9). CASE VIII A 71-year-old man with a cicatricial patch on the left ala nasi measuring 2 cm in diameter with smaller areas of erosion and pigmentation stated that he had had the lesion since childhood. He had received an electrodesiccation at 68 years of age, but the lesion recurred. A pigmented lesion was identified in photographs taken at 51 years of age (Fig. 10). DISCUSSION Basal cell epithelioma is a locally invasive but rarely metastasizing neoplasm of the skin. It has, therefore, been repeatedly debated whether the tumor is to be classified as a carcinoma or as an epithelioma. As a solution, the term basalioma has been suggested,4) but this nomenclature will not clarify the peculiar characteristics either. Although the metastasis of the tumor has been quite exceptional, recurrence after treatment has been rather frequent : In our own series of patients, many of which had received treatment followed by recurrence prior to the treatment by us, the recurrences in approximately one-third of all the cases having undergone surgical excision had taken place within 3 years after the treatment.5) Clinically, basal cell epithelioma among Japanese show prominent brownish to black pigmentation which often leads to the erroneous clinical diagnosis of malignant melanoma until histologically examined. This pigmentation is due to abundant melanin pigments produced by and stored within hypertrophic and dendritic melanocytes which are considered to be secondary components of the tumor cell nests.6) The pigmentation is more frequent in longstanding tumors, indicating that the number of melanocytes will increase with the duration of the tumor.7) The duration of the tumors, i.e. the age of the patients at onset of the tumors, is entirely dependent on the subjective histories given by the patients. However, there are tendencies towards longer duration of the tumor among the younger patients.5) The size of the tumors does not necessarily parallel the duration of the tumors. In Case IV, the size of the tumor did not exceed 1 cm in diameter after 16 years, while in Case VI, rapid growth of the tumor had occurred within 7 years despite the therapeutic attempts that had been made during the period. These data suggest that the growth rate of the basal cell epithelioma is not constant but that there is a preinvasive stage during which the growth is very slow or even stationary prior to the active and invasive stage as an epithelioma. The preinvasive stage of basal cell epithelioma is thus obvious in longstanding tumors, which are frequently pigmented,7)and it is also seen among relatively young patients.5) As early as in 1933, GLASUNOW reported that a preceding lesion had been present in 35 to 36% of 99 cases of solitary basal cell epithelioma and that in a quarter of these cases (approximately 10% of all solitary basal cell epithelioma) the lesion had derived from a lesion which had been present since birth.8) Over 90% of all basal cell epithelioma are located on the head and neck, and the dense distribution on these areas has been explained as partly due to the carcinogenic effect of sunlight on the exposed areas. However, the distribution of basal cell epithelioma differs slightly from that of squamous cell carcinoma, the development of which is apparently associated with sunlight exposure. The distribution of basal cell epithelioma on the face is most dense along the proposed lines of the

visible in photographs taken at 44, 19 and 29 years of age respectively, although this finding did not necessarily rule out the possible presence of a lesion; camera focus, print size, lesion size,

7 visible in photographs taken at 44, 19 and 29 years of age respectively, although this finding did not necessarily rule out the possible presence of a lesion; camera focus, print size, lesion size, and the degree of pigmentation have to be taken into consideration before a final conclusion as to the presence or absence of a cer- tain lesion will be made. However, at least the following minimum durations of the tumors seem to have been confirmed from the photographs: 20 years in 2 cases, 16 years in 1 case, 7 years in 2 cases, and 6 years and 3 years in 1 case each. The size of the lesion was less than 2.3 cm in diameter in 4 cases in which no previous treat- had been instituted. The rather slow ment growth as a preinvasive stage of a solitary basal cell nevus is illustrated in Fig. 1 (1-3), Fig. 2 (2 & 3), Fig. 3 (1 & 2), Fig. 6 (2), Fig. 7 (1), Fig. 8 (1) and Fig. 9 (1). Among these, Case III (Fig. 3) seemed to fully satisfy the criteria for a solitary basal cell nevus : Early onset, slow growth until shortly before present state, relatively young age of the patient, hemispheric appearance with smooth surface, no ulcerative or inflammatory changes, abundant melanin pigments, and well circumscribed basal cell nests with abundant stroma. No skeletal anomalies were found in this case. SUMMARY In reviewing available past photographs of 8 cases of longstanding, solitary basal cell epithelioma, the lesions were shown to have been present for over 16 years in 3 cases, over 6 years in 4 cases and over 3 years in a case. The presence of a preinvasive stage of solitary basal cell epithelioma was disclosed and it lead to the conclusion that such a preinvasive stage should be separated from an invasive epithelioma stage and be designated as a solitary basal cell nevus. REFERENCES 1. MICHALOWSKI, R: Naevus sebace de Jadassohn-Un etat precancereux. Dermatologica. 124: , GORLIN, R. J., YUNIS, J. J. & N. TUNA: Multiple nevoid basal cell carcinoma, odon.togenic keratocysts and skeletal anomalies: A syndrome. Acta dermat.-venereol., 43: 39-55, 1963.

8 3. SUMMERLEY, R.: Basal-cell carcinoma. An etiologic study of patients aged 45 and under with special reference to Gorlin's syndrome. Brit. J. Derm., 77: 9-15, GOTTRON, H. A.: Basaliomprobleme. Dermat. Wchnschr., 150: , MIKI, Y.: Basal cell epithelioma among Japanese. Austral. J. Dermat., 9: , MIKI, Y.: Pigmented basal cell epithelioma. II. Histological study. Med. J. Osaka Univ., 14: 13-34, MIKI, Y.: Pigmented basal cell epithelioma. I. Statistical study. Med. J. Osaka Univ., 14: 1-12, GLASUNOW, M.: Uber das Wesen and die Histogenese des sogenannten Basalzellenkrebses. Frankfurt. Ztschr. f. Path., 46: ,

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