Localized hair follicle hamartoma

Alessi, Elvio; Azzolini, A

doi:10.1111/j.1600-0560.1993.tb01277.x

Cited by 13 publications

(6 citation statements)

References 22 publications

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“…Histopathological features of multiple follicular basaloid cell aggregations identical to those observed in our case have been described in the disorder known as generalized hair follicle hamartoma, generalized trichoepithelioma, or basaloid follicular hamartoma (7). Although the histological features of the papules resembled trichoepithelioma, we chose the nomenclature of basaloid cell hamartoma because these basaloid cells were more mature than those of trichoepithelioma (8), and the adnexal neoplasms occurred together with a systemic autoimmune disease, which is different from multiple familial basaloid follicular hamartoma. Multiple basaloid cell hamartoma with alopecia and autoimmune disease was first described by Brown et al in 1969 (1) as generalized hair-follicle hamartoma associated with alopecia, aminoaciduria, and myasthenia gravis.…”

Section: Discussionsupporting

confidence: 77%

Multiple Basaloid Cell Hamartoma with Alopecia and Autoimmune Disease (Systemic Lupus Erythematosus)

Akasaka

Kon

Mihm

1996

The Journal of Dermatology

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We report a 22-year-old Japanese woman with multiple basaloid cell hamartoma with alopecia and autoimmune disease (systemic lupus erythematosus). She presented with infiltrated large annular, brown-violet, erythematous plaques with atrophic areas in the center on her right cheek, left abdomen and left knee. She also had progressive multiple follicular keratotic papules on her scalp, face, neck, and both axilla and hair loss from her scalp, eyebrow, and axilla. Serologically, rheumatoid factor (+ + +), rheumatoid arthritis hemagglutination test (x1280), and anti-nuclear antigen (x160) were positive. Histological findings of the annular lesion showed liquefaction degeneration of basal cells, lymphocytic infiltration around hair follicles and capillaries, and panniculitis with lymphoid cell infiltration, which was diagnosed as lupus erythematosus profundus. The histological findings of multiple follicular papular lesions of the scalp and neck showed aggregations of basaloid cells, partially with hair-bulb-like structures, which was diagnosed as trichoepithelioma. Taken together, the histogenesis of multiple basaloid cell hamartoma is thought to share the same basis with autoimmune disease.

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Section: Discussionsupporting

confidence: 77%

Multiple Basaloid Cell Hamartoma with Alopecia and Autoimmune Disease (Systemic Lupus Erythematosus)

Akasaka

Kon

Mihm

1996

The Journal of Dermatology

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“…Basaloid follicular hamartoma (BFH) is the term proposed by Mehregan and Baker 9 in 1985. The cases of BFH [8][9][10][11][12][13][14] are categorized into three groups: 1) generalized type with systemic autoimmune disorders; 2) linear unilateral type; and 3) solitary localized type. Types 2 and 3 are compatible to BCN and nevus comedonicus, respectively.…”

Section: Discussionmentioning

confidence: 99%

“…It is well known that hamartomas of the hair follicles show diverse clinical and histopathological appearances and many subtypes are reported. [1][2][3][4][5][6][7][8][9][10][11][12][13][14] Localized types of the hamartoma with limited differentiation include basal cell nevus, [2][3][4][5] basal cell hamartoma with follicular differentiation, 6 hamartoma in sebaceous nevus, hamartoma of sebaceous follicles 7 and basaloid follicular hamartoma. [8][9][10][11][12][13][14] We report a unique case with a localized follicular hamartoma on the head, revealed through electron microscopic and immunohistochemical studies.…”

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confidence: 99%

A case of localized follicular hamartoma: an ultrastructural and immunohistochemical study

et al. 2000

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We report the case of a 22-year-old woman with a nevoid plaque that we termed localized follicular hamartoma. The plaque was noticed at puberty on a unilateral site of the face and scalp. Clinically, it revealed numerous, skin-colored to light brown papules alone and in groups, occasionally bearing a single hair. Histologically, branched epithelial nests of squamoid and/or basaloid cells were revealed in connection with the interfollicular epidermis and the upper portions of hair follicles, of which the lower portions showed normal structures. Immunohistochemically, the epithelial nests showed the keratin expression consisted with that of the infundibular epithelium. S-100-positive cells were found in the epithelial nests and the stroma. Factor XIIIa-positive dendritic cells were numerous in adjacent to the epithelial nests. Ultrastructurally, immature melanocytes with a small number of premelanosomes and Merkel cells were found in the nests. Stromal dendritic cells showed the adherent features of the cytoplasmic processes to anchoring fibrils or basal lamina of the epithelial nests. From these findings, our case is a hamartoma, which seems to be an abortive growth of secondary hair germs with a limited differentiation to the upper follicular portion.

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“…[27][28][29] ( Figure 11); and (5) a localized and solitary type that mimics a plaque of alopecia on the scalp 27 or appears as an indurated papular plaque. [30][31][32][33][34] In our opinion, some of the cases reported as basaloid follicular hamartoma are better interpreted as trichoepitheliomas, [35][36][37] basal cell carcinoma of fibroepithelioma (Pinkus) type, 38 or infundibulocystic basal cell carcinomas. 11,39 Conversely, some cases reported as trichoepitheliomas are in our opinion examples of basaloid follicular hamartomas.…”

Section: Commentmentioning

confidence: 99%