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Clear-cell acanthoma of vermilion mucosa of lower lip
Short communications & casereports
Clear-cell acanthoma of vermilion mucosa of lower lip Stndey VETERANS
Weitmer, M.D.,* Albuquerque, ADMINISTRATION
HOSI’TTAI,
W. 11.
AND UNIVERSITY
OF NEW MEXICO
SCHOOL OF MEDICINE A 63.year-old man with clear-cell acanthoma of the vermilion mucosa of the lower lip is reported. The 143 previously documented cases, including two on the lip, have all been on the skin. The clinical and histopathologie features of clear-cell acant,homa tend to favor a benign epidermal neoplasm. Until its exact nature is known, it is suggested that clear-cell acanthoma be considered a benign localized squamous-cell lesion in which an intracellular enzyme defect has resulted in the prominent nccumuhit ion of glyeogen an11 enlargement, of the cell.
C
lear-cell acanthoma, described by Degas and associates’ in 1962, is a sharply delineated area of hyperplastic epidermis in which the Malpighian cells, except for the basal layer, are enlarged as a result of clear, glycogen-rich cytoplasm and separated by a mild degree of intercellular edema. In addition, the epidermal lesion is infiltrated with polymorphonuclear leukocytes and scattered microabscesses and has considerably decreased or absent melanogenesis and a parakeratotic surface. These features have been confirmed by electron microscopy and histochrmistry.‘, s There is a chronic inflammatory infiltrate in the dermis, and dermal appendages are almost invariably normal. Of the 104 cases reviewed by Degas and Civatte” in 1970, only two were on the lip, and both were confined to t,he skin. None of the forty atltlitional cases in the American literature was on thr lip.:%-” This article presents a brief review of clear-cell acanthoma and reports the first case occurring on t,he vermilion murnsa of the lower lip. LITERATURE
REVIEW
Clear-cell acanthoma to date has been encountered only on the skin. It typically appears as an asymptomatic solitary pink to red-brown plaque or Read at the twenty-seventh annual meeting of the American Academy of Oral Pathology, May 14 to 19, 1973, Montreal, Canada. *Assistant Chief, Laboratory Service, Veterans Administration Hospital, Albuquerque, N. M., and Associate Professor of Pathology, University of New Mexico School of Medicine.
911
912
Weitmer
Oral Surg. June, 1974
Fig. 1. Shave biopsy of lesion of the vermilion mucosa of the lower lip. Left, Clear-cell xcanthomn; right, normal squamous mucosa. (Hematosylin and eoain stain. Original magnifieation, x13.)
Fig. 2’. Higher power of Fig. 1, left, showing enlarged squamous cells with clear cytoplasm except for basal layer. The surface is covered with parakeratosis. (Hematoxylin and eosin stain. Original magnification, x40.)
nodule with a thin crust. The lesion is usually 5 to 20 mm. in diameter; most have been 10 to 15 mm. in diameter. The largest (giant form) measured 4.5 by 4.0 by 0.5 em. in greatest dimensions. There is no sex predilection. The patients have been 20 to 83 years of age; the majority were 50 to 70 years of age. The duration of the lesion before therapy varied from several months to 40 years, but in most cases it was 2 to 10 years. Of the 143 eases of clear-cell acanthoma, there were 122 (85.2 per cent) on the lower extremity including eighty-three (58 per cent) on the leg, two (1.4 per cent) on the forearm, 12 (8.4 per cent) on the trunk, and seven (5.0 per cent) on the head. Two of the latter involved the lip2 and one each the cheek, forehead, retroauricular area,2 ear,4 and nose.” Multiple lesions, all on the lower extremity, occurred in six patients2, 3 Clear-cell aeanthoma was considered or recognized in thirteen of seventy-one patients in
Volume 37 Number 6
Clear-cell
ncaxthoma
913
Fig. J. Higher power of Fig. 2 emphasizing enlarged squamous cells with clear cytoplasm and showing mild interstitial edema and several minute collections of acute inflammatory cells. (Hematoxylin and eosin stain. Original magnification, x205.)
whom the preoperative diagnosis was available. It was most often confused with basal-cell carcinoma (fifteen), histiocytoma (fourteen), pyogenic granuloma (eight), and verruca (seven), as well as nevus, malignant melanoma, Kaposi sarcoma, Bowen’s disease, and seborrheic keratosis in several instances each. CASE REPORT A 63.year-old white man underwent shave biopsy on Nov. 3, 1972, for a 9 by 7 mm., slightly elevated, rose-gray plaque on the midportion of the vermilion mucosa of the lower lip of one year’s duration. The clinical impression was leukoplakia. The specimen consisted of two portions of smooth mucosa-lined soft tissue, each measuring 5 mm. in greatest dimensions. Microscopic examination revealed prominent acanthosis of the squamous mucosa. The Malpighian cells, except for the basal layer, mere enlarged with clear cytoplasm. They were separated by a mild degree of intercellular edema infiltrated in places with few polymorphonuclear leukocytes (Figs. 1 to 3). The surface was covered with parakeratosis. The submueosa was mildly infiltrated with chronic inflammatory cells. The clear cells mere strongly PAS-positive but negative with pretreatment by diastase. The Fontana stain was negative. The adjacent mucosa was not renmrknble. The diagnosis was clear-cell acanthoma of the vermilion mucosa of the lower lip. Three months later, the patient developed a 10 l)y 7 mm. superficial white plaque in the vicinity of the previous biopsy and adjacent mucosa. Histopathologic examination of the vermilionectomy specimen obtained on March 22, 1973, disclosed mild dysplasia and cicatrix. There was no residual clear-cell acantlronm. The plastic repair of the lower lip healed ~~11. T11rw WIS no new lcnion on May 7, 1973.
DISCUSSION
Clear-cell
acanthoma has previously been (locumentctl only on the skin. Its on the \-ermilion mucosa is not surprising, since a variet!- of other cutaneous lesions occur on the lip ant1 oral mucosa. The etiology of clear-ccl1 acanthoma is unknown. Trauma is improbable, since the subsequent de\-clopmcnt~ of a clear-cell acanthoma he.s been alluded to in several instances only. Electron microscopy, cultures, and autoinoculat,ion studies have failed to disclose a virus2 or, to my knowledge, any infectious agent. In addition, there is no documentation of clear-cell acanthoma resolving in response to anti-inflammatory therapy. Most authors consider clear-cell acanthoma to represent a benign rpitlermal neoplasm. This appears to be the most likely possibility because of its clinical ant1 microscopic features: a long-standing welldemarcated, noninvasive lesion composed of essentially uniform benign-appearing Malpighian cells. Its benign nature is also attested to by the lack of evidence of progression to malignancy. The presence of reduced quantities of cytochrome osidase, succinic tlehydrogenase, and phosphorylase in clear-cell acanthoma favors epidermal origin, since sweat ducts and sweat gland neoplasms are very rich in these enzymes.” The conspicuous absence of any ectopic sweat glands or other appendages in the shave biopsy and vermilionectomy specimens in my case indicates squamous-cell origin. I believe that, until the precise nature is tletermincd, clear-cell acanthoma should hc regarded as a benign localizetl lesion of squamous-cell origin in which an intracellular enzyme defect has resulted in the prominent accumulation of glycogen and enlargement of the cell. occ~u'rcnce
REFERENCES
I. Degos, R., Delort, J., Civatte, J., and Baptiste, A. P.: Tumeur Cpidermique d’aspect particulier : Aeanthome a cellules elaires, Ann. Dermatol. Syph. (Paris) 89: 361-371, 1962. Experience of 8 Years, Br. J. Dermatol. 2. Degos, R., and Civatte, J.: Clear-cell Acanthoma: 83:
248-254.
1970.
3. Landry, M.j and Winkelmann, R. K.: Multiple Clear-cell Acanthoma and Ichthyosis, Arch. Dermatol. 105: 371-383. 1972. 4. Weitzner, S., and Harville, D. I).: Clear-cell Acanthoma of Ear: Report of a Case, Southwest. Med. 50: 96-97, 1969. 5. Brownstein, M. H.. Fernando. S.. and Shaairo. L.: Clear-cell Acanthoma : Clinicouathologic Analysis of 37 New Cases, Am. J. Clin. P&hoi. 59: 306-311, 1973. (acanthome a cellules claires). A 6. Well, G. C., and Wilson-Jones, E.: Degas’ Acanthoma Report of Five Cases With Particular Reference to the Histochemistry, I Br. J. Dermntol. 79: Eleprint
249-258, requests
1967. to :
Stanley Weitzner, M.1) Laboratory Service V. A. Hospital 2100 Ridgecrest Dr., SE Albuquerque, N. M. 87168
Clear-cell acanthoma of vermilion mucosa of lower lip Stndey VETERANS
Weitmer, M.D.,* Albuquerque, ADMINISTRATION
HOSI’TTAI,
W. 11.
AND UNIVERSITY
OF NEW MEXICO
SCHOOL OF MEDICINE A 63.year-old man with clear-cell acanthoma of the vermilion mucosa of the lower lip is reported. The 143 previously documented cases, including two on the lip, have all been on the skin. The clinical and histopathologie features of clear-cell acant,homa tend to favor a benign epidermal neoplasm. Until its exact nature is known, it is suggested that clear-cell acanthoma be considered a benign localized squamous-cell lesion in which an intracellular enzyme defect has resulted in the prominent nccumuhit ion of glyeogen an11 enlargement, of the cell.
C
lear-cell acanthoma, described by Degas and associates’ in 1962, is a sharply delineated area of hyperplastic epidermis in which the Malpighian cells, except for the basal layer, are enlarged as a result of clear, glycogen-rich cytoplasm and separated by a mild degree of intercellular edema. In addition, the epidermal lesion is infiltrated with polymorphonuclear leukocytes and scattered microabscesses and has considerably decreased or absent melanogenesis and a parakeratotic surface. These features have been confirmed by electron microscopy and histochrmistry.‘, s There is a chronic inflammatory infiltrate in the dermis, and dermal appendages are almost invariably normal. Of the 104 cases reviewed by Degas and Civatte” in 1970, only two were on the lip, and both were confined to t,he skin. None of the forty atltlitional cases in the American literature was on thr lip.:%-” This article presents a brief review of clear-cell acanthoma and reports the first case occurring on t,he vermilion murnsa of the lower lip. LITERATURE
REVIEW
Clear-cell acanthoma to date has been encountered only on the skin. It typically appears as an asymptomatic solitary pink to red-brown plaque or Read at the twenty-seventh annual meeting of the American Academy of Oral Pathology, May 14 to 19, 1973, Montreal, Canada. *Assistant Chief, Laboratory Service, Veterans Administration Hospital, Albuquerque, N. M., and Associate Professor of Pathology, University of New Mexico School of Medicine.
911
912
Weitmer
Oral Surg. June, 1974
Fig. 1. Shave biopsy of lesion of the vermilion mucosa of the lower lip. Left, Clear-cell xcanthomn; right, normal squamous mucosa. (Hematosylin and eoain stain. Original magnifieation, x13.)
Fig. 2’. Higher power of Fig. 1, left, showing enlarged squamous cells with clear cytoplasm except for basal layer. The surface is covered with parakeratosis. (Hematoxylin and eosin stain. Original magnification, x40.)
nodule with a thin crust. The lesion is usually 5 to 20 mm. in diameter; most have been 10 to 15 mm. in diameter. The largest (giant form) measured 4.5 by 4.0 by 0.5 em. in greatest dimensions. There is no sex predilection. The patients have been 20 to 83 years of age; the majority were 50 to 70 years of age. The duration of the lesion before therapy varied from several months to 40 years, but in most cases it was 2 to 10 years. Of the 143 eases of clear-cell acanthoma, there were 122 (85.2 per cent) on the lower extremity including eighty-three (58 per cent) on the leg, two (1.4 per cent) on the forearm, 12 (8.4 per cent) on the trunk, and seven (5.0 per cent) on the head. Two of the latter involved the lip2 and one each the cheek, forehead, retroauricular area,2 ear,4 and nose.” Multiple lesions, all on the lower extremity, occurred in six patients2, 3 Clear-cell aeanthoma was considered or recognized in thirteen of seventy-one patients in
Volume 37 Number 6
Clear-cell
ncaxthoma
913
Fig. J. Higher power of Fig. 2 emphasizing enlarged squamous cells with clear cytoplasm and showing mild interstitial edema and several minute collections of acute inflammatory cells. (Hematoxylin and eosin stain. Original magnification, x205.)
whom the preoperative diagnosis was available. It was most often confused with basal-cell carcinoma (fifteen), histiocytoma (fourteen), pyogenic granuloma (eight), and verruca (seven), as well as nevus, malignant melanoma, Kaposi sarcoma, Bowen’s disease, and seborrheic keratosis in several instances each. CASE REPORT A 63.year-old white man underwent shave biopsy on Nov. 3, 1972, for a 9 by 7 mm., slightly elevated, rose-gray plaque on the midportion of the vermilion mucosa of the lower lip of one year’s duration. The clinical impression was leukoplakia. The specimen consisted of two portions of smooth mucosa-lined soft tissue, each measuring 5 mm. in greatest dimensions. Microscopic examination revealed prominent acanthosis of the squamous mucosa. The Malpighian cells, except for the basal layer, mere enlarged with clear cytoplasm. They were separated by a mild degree of intercellular edema infiltrated in places with few polymorphonuclear leukocytes (Figs. 1 to 3). The surface was covered with parakeratosis. The submueosa was mildly infiltrated with chronic inflammatory cells. The clear cells mere strongly PAS-positive but negative with pretreatment by diastase. The Fontana stain was negative. The adjacent mucosa was not renmrknble. The diagnosis was clear-cell acanthoma of the vermilion mucosa of the lower lip. Three months later, the patient developed a 10 l)y 7 mm. superficial white plaque in the vicinity of the previous biopsy and adjacent mucosa. Histopathologic examination of the vermilionectomy specimen obtained on March 22, 1973, disclosed mild dysplasia and cicatrix. There was no residual clear-cell acantlronm. The plastic repair of the lower lip healed ~~11. T11rw WIS no new lcnion on May 7, 1973.
DISCUSSION
Clear-cell
acanthoma has previously been (locumentctl only on the skin. Its on the \-ermilion mucosa is not surprising, since a variet!- of other cutaneous lesions occur on the lip ant1 oral mucosa. The etiology of clear-ccl1 acanthoma is unknown. Trauma is improbable, since the subsequent de\-clopmcnt~ of a clear-cell acanthoma he.s been alluded to in several instances only. Electron microscopy, cultures, and autoinoculat,ion studies have failed to disclose a virus2 or, to my knowledge, any infectious agent. In addition, there is no documentation of clear-cell acanthoma resolving in response to anti-inflammatory therapy. Most authors consider clear-cell acanthoma to represent a benign rpitlermal neoplasm. This appears to be the most likely possibility because of its clinical ant1 microscopic features: a long-standing welldemarcated, noninvasive lesion composed of essentially uniform benign-appearing Malpighian cells. Its benign nature is also attested to by the lack of evidence of progression to malignancy. The presence of reduced quantities of cytochrome osidase, succinic tlehydrogenase, and phosphorylase in clear-cell acanthoma favors epidermal origin, since sweat ducts and sweat gland neoplasms are very rich in these enzymes.” The conspicuous absence of any ectopic sweat glands or other appendages in the shave biopsy and vermilionectomy specimens in my case indicates squamous-cell origin. I believe that, until the precise nature is tletermincd, clear-cell acanthoma should hc regarded as a benign localizetl lesion of squamous-cell origin in which an intracellular enzyme defect has resulted in the prominent accumulation of glycogen and enlargement of the cell. occ~u'rcnce
REFERENCES
I. Degos, R., Delort, J., Civatte, J., and Baptiste, A. P.: Tumeur Cpidermique d’aspect particulier : Aeanthome a cellules elaires, Ann. Dermatol. Syph. (Paris) 89: 361-371, 1962. Experience of 8 Years, Br. J. Dermatol. 2. Degos, R., and Civatte, J.: Clear-cell Acanthoma: 83:
248-254.
1970.
3. Landry, M.j and Winkelmann, R. K.: Multiple Clear-cell Acanthoma and Ichthyosis, Arch. Dermatol. 105: 371-383. 1972. 4. Weitzner, S., and Harville, D. I).: Clear-cell Acanthoma of Ear: Report of a Case, Southwest. Med. 50: 96-97, 1969. 5. Brownstein, M. H.. Fernando. S.. and Shaairo. L.: Clear-cell Acanthoma : Clinicouathologic Analysis of 37 New Cases, Am. J. Clin. P&hoi. 59: 306-311, 1973. (acanthome a cellules claires). A 6. Well, G. C., and Wilson-Jones, E.: Degas’ Acanthoma Report of Five Cases With Particular Reference to the Histochemistry, I Br. J. Dermntol. 79: Eleprint
249-258, requests
1967. to :
Stanley Weitzner, M.1) Laboratory Service V. A. Hospital 2100 Ridgecrest Dr., SE Albuquerque, N. M. 87168