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[Histopathological study of basaloid follicular hamartoma].
Ann Dermatol Venereol. 2019 Mar; 146(3):181-191.AD

Abstract

INTRODUCTION

Basaloid follicular hamartoma (BFH) is a rare tumor first described in 1985. It bears clinical and histologic similarities with basal cell carcinoma (BCC), in particular the so-called infundibulocystic form. We performed a single-center clinicopathological study of a series of typical cases of this entity that is occasionally difficult to diagnose.

MATERIALS AND METHODS

All cases of BFH seen at the Dermatopathology Laboratory of Strasbourg University Hospital were included and analyzed by means of HE staining and Ber-EP4 and PHLDA1 immunolabelling. Diagnosis was made in the event of basaloid proliferation with anastomosing cords developed from a hair follicle. Clinical data were collected from clinical files.

RESULTS

We identified 15 cases in 13 patients of mean age 44.8 years (range: 4 to 90) and the sex-ratio was 5/8. Lesions consisted of flesh-colored papules measuring 0.3 to 1.2cm in diameter, without any preferential site. Three patients had multiple lesions consisting of several coalescent papules on a breast for one, sparse papules on the back for another, and hundreds of linear unilateral BFHs, associated with osseous abnormalities, characteristic of Happle-Tinschert syndrome, for the third. All tumors were limited to the superficial and mid dermis, with a vertical orientation and connection to the epidermis in 14 of the 15 patients. In some cases, the outermost cells were basophilic while the central cells were eosinophilic. Peripheral palisading and clefting were frequently observed. Keratin cysts or sebaceous ducts were consistently present, indicating follicular differentiation. Ber-EP4+ cells were restricted to the periphery of the cords and PHLDA1 was weakly expressed.

DISCUSSION

BFH is a rare entity that must be differentiated from BCC. It presents as solitary or multiple lesions, either grouped in plaques or with a generalized or linear unilateral distribution. Generalized BFH may be associated with autoimmune diseases and linear unilateral BFH with osseous, dental and cerebral abnormalities in Happle-Tinschert syndrome. It is important to distinguish BFH from BCC to avoid inappropriate aggressive treatment.

Authors+Show Affiliations

Clinique dermatologique, université de Strasbourg, hôpitaux universitaires de Strasbourg, 1, place de l'Hôpital, 67091 Strasbourg cedex, France. Electronic address: g.hazemann@live.fr.Centre hospitalier Emile-Muller, 20, avenue du Dr-René-Laennec, 68100 Mulhouse, France.Hôpitaux civils de Colmar, 39, rue de la Liberté, 68000 Colmar, France.Clinique dermatologique, université de Strasbourg, hôpitaux universitaires de Strasbourg, 1, place de l'Hôpital, 67091 Strasbourg cedex, France.Clinique dermatologique, université de Strasbourg, hôpitaux universitaires de Strasbourg, 1, place de l'Hôpital, 67091 Strasbourg cedex, France.

Pub Type(s)

Journal Article

Language

fre

PubMed ID

30691875

Citation

Hazemann, G, et al. "[Histopathological Study of Basaloid Follicular Hamartoma]." Annales De Dermatologie Et De Venereologie, vol. 146, no. 3, 2019, pp. 181-191.
Hazemann G, Michel C, Mahé A, et al. [Histopathological study of basaloid follicular hamartoma]. Ann Dermatol Venereol. 2019;146(3):181-191.
Hazemann, G., Michel, C., Mahé, A., Lipsker, D., & Cribier, B. (2019). [Histopathological study of basaloid follicular hamartoma]. Annales De Dermatologie Et De Venereologie, 146(3), 181-191. https://doi.org/10.1016/j.annder.2018.12.007
Hazemann G, et al. [Histopathological Study of Basaloid Follicular Hamartoma]. Ann Dermatol Venereol. 2019;146(3):181-191. PubMed PMID: 30691875.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - [Histopathological study of basaloid follicular hamartoma]. AU - Hazemann,G, AU - Michel,C, AU - Mahé,A, AU - Lipsker,D, AU - Cribier,B, Y1 - 2019/01/26/ PY - 2018/04/19/received PY - 2018/06/29/revised PY - 2018/12/03/accepted PY - 2019/1/30/pubmed PY - 2019/1/30/medline PY - 2019/1/30/entrez KW - Basal cell nevus KW - Basaloid follicular hamartoma KW - Carcinome basocellulaire infundibulo-kystique KW - Gorlin syndrome KW - Hamartome folliculaire basaloïde KW - Happle-Tinschert syndrome KW - Infundibulocystic basal cell carcinoma KW - Nævus basocellulaire KW - Syndrome d’Happle-Tinschert KW - syndrome de Gorlin SP - 181 EP - 191 JF - Annales de dermatologie et de venereologie JO - Ann Dermatol Venereol VL - 146 IS - 3 N2 - INTRODUCTION: Basaloid follicular hamartoma (BFH) is a rare tumor first described in 1985. It bears clinical and histologic similarities with basal cell carcinoma (BCC), in particular the so-called infundibulocystic form. We performed a single-center clinicopathological study of a series of typical cases of this entity that is occasionally difficult to diagnose. MATERIALS AND METHODS: All cases of BFH seen at the Dermatopathology Laboratory of Strasbourg University Hospital were included and analyzed by means of HE staining and Ber-EP4 and PHLDA1 immunolabelling. Diagnosis was made in the event of basaloid proliferation with anastomosing cords developed from a hair follicle. Clinical data were collected from clinical files. RESULTS: We identified 15 cases in 13 patients of mean age 44.8 years (range: 4 to 90) and the sex-ratio was 5/8. Lesions consisted of flesh-colored papules measuring 0.3 to 1.2cm in diameter, without any preferential site. Three patients had multiple lesions consisting of several coalescent papules on a breast for one, sparse papules on the back for another, and hundreds of linear unilateral BFHs, associated with osseous abnormalities, characteristic of Happle-Tinschert syndrome, for the third. All tumors were limited to the superficial and mid dermis, with a vertical orientation and connection to the epidermis in 14 of the 15 patients. In some cases, the outermost cells were basophilic while the central cells were eosinophilic. Peripheral palisading and clefting were frequently observed. Keratin cysts or sebaceous ducts were consistently present, indicating follicular differentiation. Ber-EP4+ cells were restricted to the periphery of the cords and PHLDA1 was weakly expressed. DISCUSSION: BFH is a rare entity that must be differentiated from BCC. It presents as solitary or multiple lesions, either grouped in plaques or with a generalized or linear unilateral distribution. Generalized BFH may be associated with autoimmune diseases and linear unilateral BFH with osseous, dental and cerebral abnormalities in Happle-Tinschert syndrome. It is important to distinguish BFH from BCC to avoid inappropriate aggressive treatment. SN - 0151-9638 UR - https://www.unboundmedicine.com/medline/citation/30691875/[Histopathological_study_of_basaloid_follicular_hamartoma]_ L2 - https://linkinghub.elsevier.com/retrieve/pii/S0151-9638(18)31351-6 DB - PRIME DP - Unbound Medicine ER -