Familial multiple desmoplastic trichoepitheliomas

Shapiro, P. E.

doi:10.1001/archderm.127.1.83

Cited by 11 publications

(9 citation statements)

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“…Familial DT has been reported, with a mother and two daughters developing facial lesions, 6 as has familial multiple DT, with a mother and son with multiple scalp and facial lesions 7 . The three histological characteristics reported in original cases of DT were present in our case, supporting the diagnosis.…”

Section: Reportsupporting

confidence: 76%

Congenital desmoplastic trichoepithelioma

et al. 2007

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Desmoplastic trichoepithelioma (DT) is a rare benign adnexal neoplasm considered to have follicular differentiation. It usually presents as an asymptomatic, firm, annular plaque with a raised border. The most common site of occurrence is the face, usually on the cheek. Females are more often affected than males and the age range of patients previously reported is 8-79 years. We present a case of congenital desmoplastic trichoepithelioma. A girl was born at term to a healthy mother after an uneventful pregnancy and was noted to have widespread erythematous plaques with milia-like lesions over the right scalp, face and neck, with some areas of atrophic scarring. Histology and immunohistochemistry of incisional biopsies of the lesions were consistent with a diagnosis of DT. To our knowledge, this is the first reported case of congenital DT.

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Section: Reportsupporting

confidence: 76%

Congenital desmoplastic trichoepithelioma

et al. 2007

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“…Resolution of lesions was seen as early as three months with clearance of 80% of treated lesions. We cannot comment on how effective this treatment regimen would be on variants of trichoepitheliomas such as desmoplastic trichoepitheliomas (11) or giant solitary trichoepitheliomas (12,13). However, the advantage of using this nonsurgical treatment is no scarring, painless, and no need for other invasive procedures such as injection of local anesthetic.…”

Section: Discussionmentioning

confidence: 99%

Treatment of Multiple Trichoepitheliomas with Topical Imiquimod and Tretinoin

Urquhart

Weston

2005

Pediatric Dermatology

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An 11-year-old white girl presented multiple flesh-colored dome-shaped papules on the forehead, nose, scalp, and posterior neck. The lesions had been present for 2 years and were asymptomatic. Family history was negative for skin diseases. A skin biopsy specimen revealed histopathology consistent with trichoepitheliomas. Treatment of multiple trichoepitheliomas is usually difficult. Methods reported in the literature include laser treatment, surgery, cryotherapy, electrodessication, and radiation. The parents were concerned about the risk of scarring and wanted to pursue a nonscarring treatment. The patient was initially treated with topical imiquimod three times a week. She progressively increased the frequency of application to twice daily and added topical tretinoin gel once daily to her regimen for more resistant lesions. After 3 years of treatment, the patient experienced approximately 80% clearing of lesions without scarring. The advantage of using this nonsurgical treatment is no scarring, painless, and no need for other invasive procedures such as injection of local anesthetic.

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“…The authors noted that familial desmoplastic trichoepithelioma is extremely rare, having identified only one previous report, that by Dervan et al 2 . Dr Alfred Kopf and I also described an example of familial desmoplastic trichoepithelioma which, to the best of my knowledge, is the only extant report of familial multiple desmoplastic trichoepithelioma 3 . If one scours the literature, one can find, in the work of Ackerman et al ., 4 additional references to multiple desmoplastic trichoepithelioma inherited in an autosomal dominant fashion, but these citations are erroneous, as the report of Lazorik and Wood 5 describes multiple desmoplastic trichoepitheliomas that were not demonstrated to be familial, and the article by Ingels 6 describes a process that was reported not to be familial and that probably does not represent desmoplastic trichoepithelioma (the photomicrographs provided from this 1935 article do not allow a clear assessment of the histopathologic features, and the clinical description of the process, which describes small papules involving the entire cutaneous surface except for the palms and soles, does not seem consistent with desmoplastic trichoepithelioma).…”

mentioning

confidence: 97%

Familial desmoplastic trichoepithelioma

Philip¹

2007

Int J Dermatology

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Familial multiple desmoplastic trichoepitheliomas

Cited by 11 publications

References 0 publications

Congenital desmoplastic trichoepithelioma

Congenital desmoplastic trichoepithelioma

Treatment of Multiple Trichoepitheliomas with Topical Imiquimod and Tretinoin

Familial desmoplastic trichoepithelioma

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