Condyloma and coincidental epidermodysplasia verruciformis acanthoma positive for human papillomavirus-14 and -21.

Epidermodysplasia verruciformis (EDV) is a rare genodermatosis that predisposes individuals to persistent infection with ß-human papillomavirus (HPV) genotypes. The term EDV acanthoma may be applied to lesions with incidental findings of EDV-defining histopathological features without clinical signs of EDV. We report a case of HPV-14- and -21-positive EDV acanthoma arising in association with condyloma in a female patient with a history of low-grade squamous intraepithelial lesion of the cervix positive for high-risk HPV (non-16/18), chronic kidney disease, and systemic lupus erythematosus. The patient had no family or personal history of EDV, but the patient was on immunosuppressive therapy with mycophenolate mofetil and prednisone. A biopsy specimen from one of the perianal lesions revealed histopathologic changes consistent with EDV in the setting of condyloma. Molecular testing showed HPV-14 and -21, which supported the coexistence of condyloma with EDV acanthoma.

Response of pruritic scrotal epidermolytic acanthomas to pimecrolimus 1% cream.

Epidermolytic acanthomas (EA) are rare benign tumors of unclear etiology that present as flat, sometimes slightly keratotic, pale or whitish papules that are usually asymptomatic. Not uncommonly, their clinical appearance in the anogenital area might lead to misdiagnosis as other lesions that commonly develop at this site, such as condylomata acuminata. Though mainly asymptomatic, there are also reports of EA presenting with persistent genital pruritus. We describe the first reported case of pruritic scrotal EA successfully treated with topical pimecrolimus.

[Multiple epidermolytic acanthomas of the genitalia]. / Hyperkératose épidermolytique génitale (acanthomes épidermolytiques génitaux multiples).

BACKGROUND: Epidermolytic hyperkeratosis presents a particular histological image common to several clinical pictures, including that of keratinopathic ichthyoses. It may also occur fortuitously in various tumoral and inflammatory lesions. It is the elementary histopathological lesion of epidermolytic acanthoma, which may either be single or multiple, and when it occurs in the genital area, is known as epidermolytic hyperkeratosis of the genitalia or multiple epidermolytic acanthoma of the genitalia. Herein, we report two characteristic cases of epidermolytic hyperkeratosis of the genitalia. PATIENTS AND METHODS: The first patient was a 50-year-old woman consulting for vulvar pain in whom clinical examination revealed the presence of multiple papules on the labia majora and minora. The second patient was a 44-year-old man consulting for verrucous lesions of the scrotum. In both cases, biopsy revealed an histopathological aspect identical with acanthosis, hyperkeratosis, changes in the keratinocytes, in which the cytoplasm contained clear vacuoles, numerous keratohyalin granules and eosinophilic bodies, resulting in a diagnosis of epidermolytic hyperkeratosis of the genitalia. DISCUSSION: Epidermolytic hyperkeratosis of the genitalia is a rare disease, occurring in middle-aged men and women, but chiefly men. The lesions found on the genital organs may be either single, or, more frequently, multiple, and are described as hyperkeratotic papules, which are also reported under the term multiple epidermolytic acanthomas of the genitalia. The aetiology is unknown; certain authors incriminate a traumatic factor; the role of human papillomavirus (HPV) has been suggested but immunohistochemical studies and molecular biology studies generally reveal no viral DNA. Where lesions cause discomfort to the patient, treatment consists of emollients combined with destruction by cryotherapy or CO2 laser. Imiquimod, calcipotriol, tacrolimus and pimecrolimus have all resulted in regression of lesions.

Multiple localized epidermolytic acanthomas of the vulva associated with vulvar pruritus: a case report.

Epidermolytic acanthoma (EA) is a rare benign acquired tumor that most commonly presents as a solitary lesion on the trunk, extremities, male genitalia, and face with a male predominance. Cases occurring on the female genitalia have rarely been reported. We describe the case of multiple localized EAs occurring on the vulva of a 31-year-old woman presenting with the chief complaint of vulvar pruritus. Although rare to occur in the vulvar area, EAs should be included in the differential diagnosis of multiple vulvar papules.

Anogenital epidermolytic acanthomas: effective treatment of pruritus with 0.1% tacrolimus ointment.

Epidermolytic acanthoma is an uncommon benign tumor with a predilection for the anogenital region, that is characterized histologically by epidermolytic hyperkeratosis. Most reported cases are asymptomatic while some are pruritic. Treatment of this condition is challenging and not well studied. We report a 55-year-old man with pruritic anogenital epidermolytic acanthomas that persisted despite various topical and ablative therapies, and whose pruritus was not relieved with emollients and antihistamines. He was given 0.1% tacrolimus ointment twice daily and experienced a marked improvement in his pruritus within 2 weeks. At 1 month, there was marked improvement in itch score from 8 to 3.5 (maximum of 10), itch frequency from 20 times a day to once every 4 days, sleep score from 5 to 1 (maximum of 5), and Dermatology Life Quality Index score from 15 to 5 (maximum of 30). Apart from a transient burning sensation, no other adverse effects were noted. To our knowledge, this is the first report of the effective use of a topical calcineurin inhibitor in relieving the intractable itch associated with epidermolytic acanthoma.

Disseminated eruptive clear cell acanthoma with spontaneous regression: further evidence of an inflammatory origin?

Clear cell acanthoma (CCA) is a benign epidermal lesion with distinctive clinicopathological features. Multiple disseminated eruptive CCA is an infrequent clinical variant that has been rarely reported. It is characterized by the presence of more than 30 lesions from 1 to 10 mm in diameter that appear progressively over the years. We report the case of a 65-year-old woman with multiple disseminated eruptive CCA affecting her lower extremities. In contrast to previous reports, most of the lesions appeared in a short period of time (less than a month) and, what is more interesting is that some of them have regressed spontaneously leaving residual hyperpigmentation. At present, the histogenesis and etiology of CCA remain unknown. Accumulating data suggest a reactive origin associated with a variety of different inflammatory conditions. The case presented in this report further substantiates that CCA is indeed a reactive epidermal reaction pattern with an inflammatory etiology.

Laugier-Hunziker Syndrome Presenting with Metachronous Melanoacanthomas.

Laugier-Hunziker syndrome (LHS, also termed idiopathic lenticular mucocutaneous hyperpigmentation) is an unusual condition characterized by progressive pigmentation of the mucous membranes. LHS displays a benign course and is not associated with malignancy. Here we present a case of LHS with a 7-year follow-up. We document metachronous oral melanoacanthomas in this individual. In addition, we found that the oral melanotic macules in this patient waxed and waned in a cyclical manner. To our knowledge, this is the first report of these findings in the context of LHS. Finally, we provide an overview of other conditions that can present with mucosal hyperpigmentation. It is critical to distinguish LHS from other conditions characterized by mucosal pigmentation in order to facilitate optimal patient care.

Acantholytic dyskeratotic acanthoma: a variant of a benign keratosis.

Acantholytic acanthoma was originally described as a solitary lesion displaying histologic features of acantholysis without dyskeratosis. Solitary, non-genital lesions displaying confluent acantholysis and dyskeratosis have not been well described in the literature, clinically or histologically. We screened cases at our institution over a 6-month period and found 28 such lesions. Lesions were most often found on the trunk as a solitary papule, for which the clinical diagnosis was often basal cell carcinoma. There was a slight female predominance. Confluent acantholysis and dyskeratosis is a histologic pattern that may present as a solitary keratosis.

Oral melanoacanthoma and oral melanotic macule: a report of 8 cases, review of the literature, and immunohistochemical analysis.

Oral melanoacanthoma (MA) is a rare, benign pigmented lesion, similar to cutaneous MA, characterized by hyperplasia of spinous keratinocytes and dendritic melanocytes. The pathogenesis of oral MA remains uncertain, although its clinical behavior is suggestive of a reactive origin. The most common intraoral sites are the buccal mucosa, lip, palate and gingiva. The average age of presentation is 28 years, mainly in blacks, with a strong female predilection. The oral melanotic macule (MM) is a small, well-circumscribed brown-to-black macule that occurs on the lips and mucous membranes. The etiology is not clear and it may represent a physiologic or reactive process. The average age of presentation is 43 years, with a female predilection. A biopsy is recommended to distinguish these lesions from each other and from other oral melanocytic lesions. We depict four cases each of oral MA and MM, affecting Caucasian and Latin American mestizo patients. The clinicopathological features of these cases reflect its ample spectrum, and to the best of our knowledge, it is the first example of oral MA affecting a Caucasian boy reported in the English literature. Therefore oral MA and MM should be considered in the differential diagnosis of pigmented lesions in the oral mucosa in these populations.

Multifocal oral melanoacanthoma associated with Addison's disease and hyperthyroidism: a case report.

Oral melanoacanthoma is a mucocutaneous, pigmented, rare, benign, and probably reactive lesion. This paper reports for the first time in the literature a case of multifocal oral melanoacanthoma in a patient diagnosed with Addison's disease and concomitant Graves' disease with hyperthyroidism. The patient presented with oral pigmented lesions, which were hypothesized to be mucosal pigmentation associated with Addison's disease. Due to their unusual clinical pattern, these oral lesions were biopsied and diagnosed as oral melanoacanthoma on histopathology and immunohistochemistry for HMB-45. At the moment of this report, the patient was being treated for her systemic conditions, but the lesions had not regressed. Reactive hyperpigmentation of the skin and mucous membranes may be found in Addison's disease and hyperthyroidism. This case reinforces the hypothesis of a reactive nature for oral melanoacanthoma and highlights the need for investigation of endocrine disorders in patients with multifocal oral melanoacanthoma.

Dermatology clinics: what's new in dermatopathology: news in nonmelanocytic neoplasia.

This article reviews the recent dermatopathology literature involving nonmelanocytic neoplasia, with a focus on important work done over the last 5 years. The discussion includes advances in the understanding of Merkel cell carcinoma pathogenesis and prognosis; changes in the seventh edition of the American Joint Committee of Cancer staging manual in reference to staging of squamous cell carcinoma and Merkel cell carcinoma; newly described or rare histopathologic patterns and entities including squamoid eccrine ductal carcinoma, rippled-pattern adnexal neoplasms, onychomatricoma, spindle cell predominant trichodiscoma/neurofollicular hamartoma, and myoepithelioma; and microsatellite instability in sebaceous neoplasms of Muir-Torre syndrome and other tumors.

Multifocal oral melanoacanthoma associated with Addison's disease and hyperthyroidism: a case report

SUMMARY Oral melanoacanthoma is a mucocutaneous, pigmented, rare, benign, and probably reactive lesion. This paper reports for the first time in the literature a case of multifocal oral melanoacanthoma in a patient diagnosed with Addison's disease and concomitant Graves' disease with hyperthyroidism. The patient presented with oral pigmented lesions, which were hypothesized to be mucosal pigmentation associated with Addison's disease. Due to their unusual clinical pattern, these oral lesions were biopsied and diagnosed as oral melanoacanthoma on histopathology and immunohistochemistry for HMB-45. At the moment of this report, the patient was being treated for her systemic conditions, but the lesions had not regressed. Reactive hyperpigmentation of the skin and mucous membranes may be found in Addison's disease and hyperthyroidism. This case reinforces the hypothesis of a reactive nature for oral melanoacanthoma and highlights the need for investigation of endocrine disorders in patients with multifocal oral melanoacanthoma.

Multifocal cutaneous melanoacanthoma with ulceration: a case report with review of literature.

We report a case of a 58-year-old female patient who presented with multiple, asymptomatic, slowly-growing, raised pigmented lesions all over her body for the past 10 years with ulceration in one of the lesions on the trunk for the past five months. Histopathology of the lesion revealed features consistent with melanoacanthoma. Here, we report the first case of cutaneous melanoacanthoma presenting with an ulcerated plaque and the third case of cutaneous melanoacanthoma with multiple lesions. To the best of our knowledge, ulceration has not yet been reported as a feature of cutaneous melanoacanthoma in the medical literature.