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Giant clear cell acanthoma in an atypical location
Volume 21 Number 2, Part 1 August 1989
lar area without an obvious cause. The name mira en plaque was suggested to describe this unusual condition. W e rePort a third case of milia en plaque. Case report. A 39-year-old Filipino woman had a slightly pruritic lesion of 1 year's duration located in the left inferior postauricular area. She denied any family history of similar lesions or of right postauricular involvement. There was no history of head or neck irradiation. The patient did not wear glasses and denied application of creams, oils, soaps, tars, or steroids to the affected area. She discontinued application of perfume to the postauricular areas soon after noticing the lesion. Physical examination showed a 1 • 3 cm erythematous plaque composed of numerous white and yellow globular bodies and open comedones (Fig. 1). Sebaceous hyperplasia was noted in the frontal and periocular areas. A biopsy specimen revealed numerous epidermal keratin-filled cysts that were consistent with milia (Fig. 2). Treatment with 0.05% tretinoin, topically administered twice daily for 3 months, resulted in resolution.
Discussion. Comedones are keratinous plugs in dilated pilosebaceous orifices and are essential comPonents of acne.1 Milia are keratinized cysts that may arise from the infundibulum of vellus hair follicles, sebaceous glands, or eccrine sweat ducts. 2 Closed comedones m a y resemble milia clinically and m a y even be associated with milia in acne.3 The rapid localized adult onset, lack of family history, and location in our patient preclude the diagnosis of any nevoid or familial comedo anomaly. Nodular elastosis with cysts and comedones (Favre-Racouchot syndrome) is unlikely because of the patient's age, sex, and site of involvement. Secondary causes of comedo formation, such as systemic or local treatment, could not be identified. Clinical and histologic findings were more consistent with the keratin cyst of milia than with the follicular plugging seen in comedones. The clinical history of this patient was similar to that of the other two reported cases. T h e cause of milia en plaque is unknown. In our case, perfumes were the only topical agents applied to the postauricular areas. Perfume is unlikely to be the cause because involvement was unilateral, despite bilateral application. Other topical agents, such as steroids 4 and fluorouracil, 5 have been implicated in causing milia. Neither agent was used by our patient. Oral administration of benoxaprofen, a 5-1ipoxygenase inhibitor, has been shown to induce milia. 6 Our patient denied use of this agent or of any nonsteroidal anti-intlammatory agents. Local treatment with topical tretinoin has been successful in all three cases.
Brief communications
313
4. Tsuji T, Kadoya A, Tanaka R, et al. Milia induced by corticosteroids. Arch Dermatol 1986;122:139-40. 5. Brenner S, Shohet J, Krakowski A, et al. Mucoid milia [Letter]. Arch Dermatol 1984;120:300. 6. Kragballe K, Herlin T. Benoxaprofen improves psoriasis: a double-blind study. Arch Dermatol 1983;1/9:548-52.
Giant clear cell acanthoma in an atypical location J. A. A. Langtry, MB, M R C P , ~ H. Torras, MD, b J. Palou, MD, b M. Lecha, M D , b and J. M. Mascaro, M D b Durham, England, and
Barcelona, Spain Clear cell acanthoma was first described by Degos et al. j in 1962. There have been numerous reports of this benign t u m o r ) including 12 cases of multiple lesions ~ From the Departments of Dermatology, Dryburn Hospital, Durham, England,~ and Hospital Clinico, Barcelona, Spain? Reprint requests: Prof. J. M. Mascaro, Department of Dermatology, Universidad de Barcelona, 08036 Barcelona, Spain.
REFERENCES 1. Hubler WR, Rudolph AH, Kelleher RM. Milia en plaque. Cutis 1978;22:67-70. 2. Tsuji T, Sugal T, Suzuki S. The mode of eecrine duct milia. J Invest Dermatol 1975;65:388-93. 3. Becker SW, Obemayer MF. Diseases of the sweat gland. In: Modern dermatology and syphilology. 2nd ed. Philadelphia: JB Lippincott, 1947:596.
Fig. 1. Irregular, erythematous plaques on medial aspect of the left buttock.
314
Journal of the American Academy of Dermatology
Brief communications
Fig. 2. Photomicrograph of giant clear cell acanthoma. Note elongated rete ridges and large, well-defined epidermal cells. (Hematoxylin-eosin stain; •
/
Fig. 3. Photomicrograph showing large, clear epidermal cells filled with labile amylase material. (Periodic acid--Schiff stain; x20.)
and a case of giant clear cell acanthoma. 4 We report the case of a patient with the giant form in an atypical location. In June 1987 an 87-year-old woman was first seen at the Hospital Clinieo y Provincial, Barcelona, with a slowly enlarging plaque on the left buttock (Fig. 1). The plaque had been present for 2 years. An irregular, erythematous plaque with a shiny, velvety surface, central elevation, and a sharply defined border was situated on the medial aspect of the left buttock (Fig. I). A similar smaller lesion was in close proximity. These lesions measured 40 • 40 mm and 10 • 12 ram, respectively. Microscopic examination of the lesion tissue revealed
a sharply demarcated acanthoma with elongated rete ridges and large, clear epidermal cells (Fig. 2). There was slight spongiosis, and numerous neutrophils were found throughout the acanthotic epidermis. There was a moderately severe, predominantly lymphocytic infiltrate. Periodic acid-Schiff reagent indicated the presence of labile amylase material filling the large, clear epidermal cells (Fig. 3). Tumors were successfully removed by curettage and electrocoagulation. Clear cell acanthoma usually occurs on the legs but has been reported to be found on other regions of the body. 5'6 It is usually solitary and varies in size from 3 to 20 mm in diameter. Duperrat et al. 4 reported a giant clear cell acanthoma 45 • 40 mm in size on the leg. The
Volume 21 Number 2, Part 1 August 1989
appearance of clear cell acanthoma in our patient was histologically typical but unusual in its size and location.
Brief communications
315
Successful treatment of the acne of Apert syndrome with isotretinoin Dixon Robison, MD, and N a n c y Anderson Wilms, M D
REFERENCES
Loma Linda, California
1. Degos R, Delort J, Civatte J, et al. Acanthome cdlules claires. Ann Dermatol Syphil (Paris) 1962;89: 361. 2. Cotton DW, Mills PW, Stephenson T J, et al. On the nature of clear cell acanthomas. Br J Dermato[ 1987; 117:569-574. 3. Baden T J, Woodley DT, Wheeler CE Jr. Multiple clear cell acanthomas. J AM ACAD D~P,MATOL1987;16:1075-8. 4. Duperrat B, Vanbremeersch F, David V, et al. Forme g~ant de l'aeanthome de Degos. Bull Soc Fr Syphil 1966; 73:884-6. 5. Degos R, Civatte J. Clear cell acanthoma. Experience of eight years. Br J Dermatol 1970;83:248-54. 6. Parhizgor B, Wood MG. Pale cell acanthoma of the scrotum. Curls 1982;30:231-3.
Acrocephalosyndactyly(Apert syndrome) is a descriptive term proposed by Apert in 1906, ~ after he observed similar characteristic anatomic deformities in several children. Anomalies of most organ systems have been described since that time. 2 The acneiform lesions were From Loma Linda University. Reprint requests: Nancy Anderson Wilms, MD, Loma Linda University Faculty Medical Offices,Suite 2600, Loma Linda, CA 92350.
Figs. 1 and 2. Severe cystic aene before isotretinoin therapy. Figs. 3 and 4. Marked resolution of cystic acne at week 16 of isotretinoin therapy.
lar area without an obvious cause. The name mira en plaque was suggested to describe this unusual condition. W e rePort a third case of milia en plaque. Case report. A 39-year-old Filipino woman had a slightly pruritic lesion of 1 year's duration located in the left inferior postauricular area. She denied any family history of similar lesions or of right postauricular involvement. There was no history of head or neck irradiation. The patient did not wear glasses and denied application of creams, oils, soaps, tars, or steroids to the affected area. She discontinued application of perfume to the postauricular areas soon after noticing the lesion. Physical examination showed a 1 • 3 cm erythematous plaque composed of numerous white and yellow globular bodies and open comedones (Fig. 1). Sebaceous hyperplasia was noted in the frontal and periocular areas. A biopsy specimen revealed numerous epidermal keratin-filled cysts that were consistent with milia (Fig. 2). Treatment with 0.05% tretinoin, topically administered twice daily for 3 months, resulted in resolution.
Discussion. Comedones are keratinous plugs in dilated pilosebaceous orifices and are essential comPonents of acne.1 Milia are keratinized cysts that may arise from the infundibulum of vellus hair follicles, sebaceous glands, or eccrine sweat ducts. 2 Closed comedones m a y resemble milia clinically and m a y even be associated with milia in acne.3 The rapid localized adult onset, lack of family history, and location in our patient preclude the diagnosis of any nevoid or familial comedo anomaly. Nodular elastosis with cysts and comedones (Favre-Racouchot syndrome) is unlikely because of the patient's age, sex, and site of involvement. Secondary causes of comedo formation, such as systemic or local treatment, could not be identified. Clinical and histologic findings were more consistent with the keratin cyst of milia than with the follicular plugging seen in comedones. The clinical history of this patient was similar to that of the other two reported cases. T h e cause of milia en plaque is unknown. In our case, perfumes were the only topical agents applied to the postauricular areas. Perfume is unlikely to be the cause because involvement was unilateral, despite bilateral application. Other topical agents, such as steroids 4 and fluorouracil, 5 have been implicated in causing milia. Neither agent was used by our patient. Oral administration of benoxaprofen, a 5-1ipoxygenase inhibitor, has been shown to induce milia. 6 Our patient denied use of this agent or of any nonsteroidal anti-intlammatory agents. Local treatment with topical tretinoin has been successful in all three cases.
Brief communications
313
4. Tsuji T, Kadoya A, Tanaka R, et al. Milia induced by corticosteroids. Arch Dermatol 1986;122:139-40. 5. Brenner S, Shohet J, Krakowski A, et al. Mucoid milia [Letter]. Arch Dermatol 1984;120:300. 6. Kragballe K, Herlin T. Benoxaprofen improves psoriasis: a double-blind study. Arch Dermatol 1983;1/9:548-52.
Giant clear cell acanthoma in an atypical location J. A. A. Langtry, MB, M R C P , ~ H. Torras, MD, b J. Palou, MD, b M. Lecha, M D , b and J. M. Mascaro, M D b Durham, England, and
Barcelona, Spain Clear cell acanthoma was first described by Degos et al. j in 1962. There have been numerous reports of this benign t u m o r ) including 12 cases of multiple lesions ~ From the Departments of Dermatology, Dryburn Hospital, Durham, England,~ and Hospital Clinico, Barcelona, Spain? Reprint requests: Prof. J. M. Mascaro, Department of Dermatology, Universidad de Barcelona, 08036 Barcelona, Spain.
REFERENCES 1. Hubler WR, Rudolph AH, Kelleher RM. Milia en plaque. Cutis 1978;22:67-70. 2. Tsuji T, Sugal T, Suzuki S. The mode of eecrine duct milia. J Invest Dermatol 1975;65:388-93. 3. Becker SW, Obemayer MF. Diseases of the sweat gland. In: Modern dermatology and syphilology. 2nd ed. Philadelphia: JB Lippincott, 1947:596.
Fig. 1. Irregular, erythematous plaques on medial aspect of the left buttock.
314
Journal of the American Academy of Dermatology
Brief communications
Fig. 2. Photomicrograph of giant clear cell acanthoma. Note elongated rete ridges and large, well-defined epidermal cells. (Hematoxylin-eosin stain; •
/
Fig. 3. Photomicrograph showing large, clear epidermal cells filled with labile amylase material. (Periodic acid--Schiff stain; x20.)
and a case of giant clear cell acanthoma. 4 We report the case of a patient with the giant form in an atypical location. In June 1987 an 87-year-old woman was first seen at the Hospital Clinieo y Provincial, Barcelona, with a slowly enlarging plaque on the left buttock (Fig. 1). The plaque had been present for 2 years. An irregular, erythematous plaque with a shiny, velvety surface, central elevation, and a sharply defined border was situated on the medial aspect of the left buttock (Fig. I). A similar smaller lesion was in close proximity. These lesions measured 40 • 40 mm and 10 • 12 ram, respectively. Microscopic examination of the lesion tissue revealed
a sharply demarcated acanthoma with elongated rete ridges and large, clear epidermal cells (Fig. 2). There was slight spongiosis, and numerous neutrophils were found throughout the acanthotic epidermis. There was a moderately severe, predominantly lymphocytic infiltrate. Periodic acid-Schiff reagent indicated the presence of labile amylase material filling the large, clear epidermal cells (Fig. 3). Tumors were successfully removed by curettage and electrocoagulation. Clear cell acanthoma usually occurs on the legs but has been reported to be found on other regions of the body. 5'6 It is usually solitary and varies in size from 3 to 20 mm in diameter. Duperrat et al. 4 reported a giant clear cell acanthoma 45 • 40 mm in size on the leg. The
Volume 21 Number 2, Part 1 August 1989
appearance of clear cell acanthoma in our patient was histologically typical but unusual in its size and location.
Brief communications
315
Successful treatment of the acne of Apert syndrome with isotretinoin Dixon Robison, MD, and N a n c y Anderson Wilms, M D
REFERENCES
Loma Linda, California
1. Degos R, Delort J, Civatte J, et al. Acanthome cdlules claires. Ann Dermatol Syphil (Paris) 1962;89: 361. 2. Cotton DW, Mills PW, Stephenson T J, et al. On the nature of clear cell acanthomas. Br J Dermato[ 1987; 117:569-574. 3. Baden T J, Woodley DT, Wheeler CE Jr. Multiple clear cell acanthomas. J AM ACAD D~P,MATOL1987;16:1075-8. 4. Duperrat B, Vanbremeersch F, David V, et al. Forme g~ant de l'aeanthome de Degos. Bull Soc Fr Syphil 1966; 73:884-6. 5. Degos R, Civatte J. Clear cell acanthoma. Experience of eight years. Br J Dermatol 1970;83:248-54. 6. Parhizgor B, Wood MG. Pale cell acanthoma of the scrotum. Curls 1982;30:231-3.
Acrocephalosyndactyly(Apert syndrome) is a descriptive term proposed by Apert in 1906, ~ after he observed similar characteristic anatomic deformities in several children. Anomalies of most organ systems have been described since that time. 2 The acneiform lesions were From Loma Linda University. Reprint requests: Nancy Anderson Wilms, MD, Loma Linda University Faculty Medical Offices,Suite 2600, Loma Linda, CA 92350.
Figs. 1 and 2. Severe cystic aene before isotretinoin therapy. Figs. 3 and 4. Marked resolution of cystic acne at week 16 of isotretinoin therapy.