Oral focal acantholytic dyskeratosis (warty dyskeratoma)

Oral focal acantholytic dyskeratosis (warty dyskeratoma) Report

of two

cases

John I,. Qiwtrc, D.X.D., U.S.,* I,. S. A. Gmez, D.X.D.,“* 0. Greer, D.D.X., Sc.D.,*** Bostoll, Il1as.s. DEPARTBIEXTS MEDICISE

OF AXD

ORAL

BOSTON

PATHOLOGY, UNIVERSITY

TUFTS SCHOOL

UNI\-ERSITY OF

GRADUATE

mcl Robed

SCHOOL

OF

DESTAL

DENTISTRY

Two cases of intraoral focal acantholytic dyskeratosis are presented. These uncommon, small, nodular, keratotic lesions, with a distinct histologic pattern similar to that of Darier’s disease, have beeu reported as warty tlyskeratomas. Treatment is conservative surgical removal and folloxv-up. It is suggested that the histologic diagnostic term of focal c~nntholytic ayskertmis he used, since there may he several clinical entities with a similar histology but different etiology.

S

mall, nodular, crateriform, keratotic lesions with a distinctive histologic pattern similar to that of Ijarier’s disease have been reportecl in the oral cavit) and have been called warty dyskeratomas or isolated dgskeratosis follicularis.], ’ These lesions are quite rare, and it is not known what causes them. Treatmrnt is conseryativc surgical removal and continued follow-up. Ackerman” has introduced the terminology of’ focal acanthol,vtie tlyskcratosis to describe a distinctive histologic pattern which he demonstrated can bc seen in several different clinical expressions, including Ijarier’s discasc. On the basis of his findings on cutaneous lesions, he has offered a classification based on size, number of lesions, and tluration. He observrtl focal a~untholytic tlyskrra(a) inapparent, (b) JMpdilI”, 01 tosis in (1 ) single lesions that were clinically *As&taut Professor, Deprtnwnt of Oral Patllology, Tufts Uuirrrsity School of Dental Mwlieine. “*Clinical Tnstructor, Department of Oral I’athology, lhrvard School of Dental Mrtlicine. **‘Formerly Special NIH Kesenrcll Fellow, l)el~artment of Oral Pathology, lioston IJniversity School of Graduate Dentistry. At prcwnt Assistant Professor and Head of Surgical Pathology, School of Dentistry, IJnivrrsitg of Colorado Mwlicxl (‘enter. 474

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Pig. 1. Case 1. A, Focal aeantholytic dyskeratosis. Oblique cut demonstrating nodularity of lesion and central cystlike crater lined with keratin. (Hematoxylin and eosin stain. Magnification, x60.) 73, Hyperorthokeratotic lining of crater, suprabasilar clefting, and acantholytic cells (IIematoxylin and eosin stain. Magnification, x200.)

(c) nodular and in (2) multiple lesions that were (a) persistent or (b) evanescent. This classification avoids etiologic factors. It is the purpose of this article to report on two cases of single oral lesions with focal acantholytic dyskeratosis, consistent with warty dyskeratoma or isolated dyskeratosis follicularis. CASE REPORT5 CASE 1

A 1972. lesion by a

51.year-old white woman consulted her dentist On examination, an asymptomatic, white-gray, was noted on the buccal mucosa opposite the upper central, depressed, craterlike area surrounded by

for routine treatment in September, circumscribed, firm, slightly nodular left cuspid area. It was characterized radiating white striae. Past medical

476

Giunta,

Gomez,

and

Oral Surg. March, 1975

Grew

C, Connective tissue papillae (villi) Fig. 1 (Cont’d). cells; note dyskeratotic and aeantholytic cells. (Hematoxylin x450. J

lined with hyperchromatic basal and eosin stain. Magnification,

history was noncontributory. There were no skin lesions. The patient had an old denture, a history of cheek biting, and smoked and drank heavily. The lesion was asymptomatic, and the duration was unknown. The lesion was excised with the patient under local anesthesia. Examination of the biopsy specimen revealed an oblique tangential cut of mucosa presenting a central depression with keratin. There were intraepithelial clefts containing acantholytic and dyskeratotic cells. The base of the clefts contained villous projecfions of connective tissue lined with an intact row of cuboidal or columnar basilar cells with dysplastic changes. The underlying tissue was infiltrated with lymphocytes, plasma cells, and histiocytes. Diagnosis

The lesion was diagnosed as warty dyskeratoma (isolated dyskeratosis follieularis). A representative section was submitted for consultation to Dr. Walter F. Lever, Professor and Chairman, Department of Dermatology, New England Medical Center Hospital. His opinion favored a diagnosis of pemphigus. However, the patient had and has had no other lesions on the mueosa or the skin, and there has been no recurrence after 2 years. CASE 2

A 33.year-old white woman was referred to a periodontist for routine periodontal evaluation in June, 1972. The patient was a cigarette smoker. Further history revealed no systemic problems. Intraoral examination revealed mild gingivitis and a single gray-white papular lesion, 3 mm. in diameter, on the anterior hard palate. The lesion was asymptomatic, and its duration was unknown. The patient denied having lesions on any other part of the body, and an examination of the visible skin areas was negative for other lesions. The lesion was excised under local anesthesia. Pathologic

Andings

Histologic examination showed moderate hyperkeratosis and acanthosis. basilar lacunae containing so-called corps ronds and grains were present.

Cleftlike

supra-

Diagnosis

The findings were consistent with a diagnosis of warty dyskeratoma (isolated dyskeratosis follicularis). A representative section was submitted for consultation to Dr. Herbert Mescon, Professor and Chairman, Department of Dermatology, Boston University School of Medicine,

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Fig. d. Case 2. A, Focal aeantholytic dyskeratosis. Keratin core, suprabasilar cleft about preserved papillae. (Hematoxylin and eosin stain. Magnification,, x200.) B, Preserved papillae, dyskeratotic and acantholytic cells. (Hematoxylin and eosin stam. Magnification, x450.)

who concurred with the diagnosis. other lesions have developed.

There has been no recurrence

in more than 2 years, and no

DISCUSSION

The number of reported cases of isolated nodular lesions in the oral cavity showing acantholytie changes is small (six, including those reported here), and no definitive statements can be made about them. However, some observations may be appropriate. It is of interest that, clinically, the lesions have been small (generally 5 mm.) nodular masses with a central depression filled with keratin. Most of them have occurred on mucosa overlying bone. However, in our Case 1 the lesion was

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definitely on unattached mucosa, which casts doubt on the proposal* that these lesions are site-specific for attached mucosa. A review of the patients’ histories reveals that the use of tobacco may be a contributing factor in the pathogenesis of these lesions. Heavy cigarette smoking or use of snuff was mentioned in four of six cases. Since tobacco usage is implicated in other keratotic lesions, it is plausible that it may be a factor in these lesions. None of the known intraoral lesions have been associated with a pilosebaceous apparatus, as have the cutaneous warty dyskeratomas. This does not preclude the existence of the oral warty dyskeratoma, as Tomich and Burkes2 have stated. However, oral and cutaneous lesions may be related only by a common histologic pattern and not by common etiology. Thus far, the other oral lesion that has been reported with a similar histologic pattern is that of Darier’s disease. In Darier’s disease, though, there are multiple persistent lesions on both the skin and the mucosa. An excellent discussion of the histologic differential diagnosis of Darier’s disease is presented by Weathers and DriscolL4 Although pemphigus may be considered, the oral lesions of pemphigus are usually multiple, not discrete, and not keratotic. It may be that there are many oral conditions that present histologically with the distinct pattern that Ackerman3 has called “focal acantholytie dyskeratosis” of the skin. According to his classification, oral lesions with a similar histologic pattern are the single, nodular lesions (warty dyskeratoma) and the multiple, persistent lesions (Darier’s disease). A search of oral biopsy services may reveal other clinical expressions of this distinct histologic pattern. In that light, the term oral focal acuntholytic dyskeratosis is presented to suggest a clinical perspective for oral lesions with common histologic findings. The term dyskera.tosis as used in this article refers merely to the histologic pattern of the cells and not to the clinical behavior or prognosis of the lesion. It does not imply that the warty dyskeratoma is a premalignant lesion. On the contrary, it appears from the cases reported that oral focal acantholytic dyskeratosis is not premalignant or related to the so-called “dyskeratotic leukoplakia.” Rather, the prognosis is good, as the lesions respond well to conservative surgical therapy and apparently do not recur. REFERENCES

A Note Concerning Its Occurrence 1. Gorlin, R. J., and Peterson, N. C.: Warty Dyskeratoma: in the Oral Mueosa, Arch. Dermatol. 95: 292-293, 1967. 2. Tomich. C. E.. and Burkes, D. J.: Wartv Dvskeratoma (Isolated Dyskeratosis Follicularis) of the bra1 Mucosa, ORAL BURG. 31: 798.-807; 1971. 3. Ackerman, A. B.: Focal Acantholytic Dyskeratosis, Arch. Dermatol. 106: 702-706, 1972. 4. Weathers, D. R., and Driscoll, R. M.: Darier’s Disease of the Oral Mimosa; Report of Five Cases, ORAL SURG. 37: 711-721, 1974. Reprint requests to: Dr. John Giunta Department of Oral Pathology Tufts University School of Dental 1 Kneeland St. Boston, Mass. 02111

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