Basaloid follicular hamartoma of the eyelid: a case report and literature review about an unusual lesion in the ocular region

Detalhes bibliográficos
Autor(a) principal: Lamas, Nuno Jorge
Data de Publicação: 2022
Outros Autores: Rodrigues, Ana Patrícia, Araújo, Maria, Vizcaíno, José Ramón, Coelho, André
Tipo de documento: Artigo
Idioma: eng
Título da fonte: Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)
Texto Completo: https://hdl.handle.net/1822/78289
Resumo: Basaloid follicular hamartoma (BFH) is a normally benign, uncommon, malformative lesion involving the hair follicles, which usually poses challenges in the differential diagnosis with other benign and malignant tumours, especially basal cell carcinoma, due to significant clinical and morphological overlap. Here, we report the case of a 53-year-old male who presented with a mass in the upper left eyelid evolving for one year. The patient had a previous history of total colectomy and an abdominal desmoid tumour within the context of Familial Adenomatous Polyposis (FAP), with a documented germline mutation in the Adenomatous Polyposis Coli (APC) gene. The eyelid lesion was biopsied and the histological analysis of the three small tissue fragments received revealed fragments with cutaneous–conjunctival lining displaying a subepithelial proliferation of basaloid nests with peripheral palisading, compatible with primitive hair follicles. There were images of anastomosis between different basaloid nests, which had their connection to the epithelial lining preserved. The stroma had high cellularity and sometimes primitive mesenchymal papillae were evident. Pleomorphism was absent, mitotic figures were barely identified, and no necrosis was seen. The basaloid nests did not have epithelial–stromal retraction nor mucin deposits. A diagnosis of BFH was proposed, which was later confirmed after surgical excision of the whole eyelid lesion. No evidence of carcinoma was present. This case illustrates the main features of the rare benign eyelid BFH. The standard medical or surgical approach of these lesions remains to be firmly established. Nearly nine months after surgical excision our patient remains well without signs of disease recurrence.
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spelling Basaloid follicular hamartoma of the eyelid: a case report and literature review about an unusual lesion in the ocular regionBasaloid follicular hamartomaEyelidEyelid tumourOcular pathologyOcular regionScience & TechnologyBasaloid follicular hamartoma (BFH) is a normally benign, uncommon, malformative lesion involving the hair follicles, which usually poses challenges in the differential diagnosis with other benign and malignant tumours, especially basal cell carcinoma, due to significant clinical and morphological overlap. Here, we report the case of a 53-year-old male who presented with a mass in the upper left eyelid evolving for one year. The patient had a previous history of total colectomy and an abdominal desmoid tumour within the context of Familial Adenomatous Polyposis (FAP), with a documented germline mutation in the Adenomatous Polyposis Coli (APC) gene. The eyelid lesion was biopsied and the histological analysis of the three small tissue fragments received revealed fragments with cutaneous–conjunctival lining displaying a subepithelial proliferation of basaloid nests with peripheral palisading, compatible with primitive hair follicles. There were images of anastomosis between different basaloid nests, which had their connection to the epithelial lining preserved. The stroma had high cellularity and sometimes primitive mesenchymal papillae were evident. Pleomorphism was absent, mitotic figures were barely identified, and no necrosis was seen. The basaloid nests did not have epithelial–stromal retraction nor mucin deposits. A diagnosis of BFH was proposed, which was later confirmed after surgical excision of the whole eyelid lesion. No evidence of carcinoma was present. This case illustrates the main features of the rare benign eyelid BFH. The standard medical or surgical approach of these lesions remains to be firmly established. Nearly nine months after surgical excision our patient remains well without signs of disease recurrence.Multidisciplinary Digital Publishing Institute (MDPI)Universidade do MinhoLamas, Nuno JorgeRodrigues, Ana PatríciaAraújo, MariaVizcaíno, José RamónCoelho, André2022-01-072022-01-07T00:00:00Zinfo:eu-repo/semantics/publishedVersioninfo:eu-repo/semantics/articleapplication/pdfhttps://hdl.handle.net/1822/78289engLamas, N.J.; Rodrigues, A.P.; Araújo, M.; Vizcaíno, J.R.; Coelho, A. Basaloid Follicular Hamartoma of the Eyelid: A Case Report and Literature Review about an Unusual Lesion in the Ocular Region. Diagnostics 2022, 12, 140. https://doi.org/10.3390/diagnostics120101402075-441810.3390/diagnostics12010140140https://www.mdpi.com/2075-4418/12/1/140info:eu-repo/semantics/openAccessreponame:Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)instname:Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informaçãoinstacron:RCAAP2023-07-21T12:49:05Zoai:repositorium.sdum.uminho.pt:1822/78289Portal AgregadorONGhttps://www.rcaap.pt/oai/openaireopendoar:71602024-03-19T19:47:29.189088Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos) - Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informaçãofalse
dc.title.none.fl_str_mv Basaloid follicular hamartoma of the eyelid: a case report and literature review about an unusual lesion in the ocular region
title Basaloid follicular hamartoma of the eyelid: a case report and literature review about an unusual lesion in the ocular region
spellingShingle Basaloid follicular hamartoma of the eyelid: a case report and literature review about an unusual lesion in the ocular region
Lamas, Nuno Jorge
Basaloid follicular hamartoma
Eyelid
Eyelid tumour
Ocular pathology
Ocular region
Science & Technology
title_short Basaloid follicular hamartoma of the eyelid: a case report and literature review about an unusual lesion in the ocular region
title_full Basaloid follicular hamartoma of the eyelid: a case report and literature review about an unusual lesion in the ocular region
title_fullStr Basaloid follicular hamartoma of the eyelid: a case report and literature review about an unusual lesion in the ocular region
title_full_unstemmed Basaloid follicular hamartoma of the eyelid: a case report and literature review about an unusual lesion in the ocular region
title_sort Basaloid follicular hamartoma of the eyelid: a case report and literature review about an unusual lesion in the ocular region
author Lamas, Nuno Jorge
author_facet Lamas, Nuno Jorge
Rodrigues, Ana Patrícia
Araújo, Maria
Vizcaíno, José Ramón
Coelho, André
author_role author
author2 Rodrigues, Ana Patrícia
Araújo, Maria
Vizcaíno, José Ramón
Coelho, André
author2_role author
author
author
author
dc.contributor.none.fl_str_mv Universidade do Minho
dc.contributor.author.fl_str_mv Lamas, Nuno Jorge
Rodrigues, Ana Patrícia
Araújo, Maria
Vizcaíno, José Ramón
Coelho, André
dc.subject.por.fl_str_mv Basaloid follicular hamartoma
Eyelid
Eyelid tumour
Ocular pathology
Ocular region
Science & Technology
topic Basaloid follicular hamartoma
Eyelid
Eyelid tumour
Ocular pathology
Ocular region
Science & Technology
description Basaloid follicular hamartoma (BFH) is a normally benign, uncommon, malformative lesion involving the hair follicles, which usually poses challenges in the differential diagnosis with other benign and malignant tumours, especially basal cell carcinoma, due to significant clinical and morphological overlap. Here, we report the case of a 53-year-old male who presented with a mass in the upper left eyelid evolving for one year. The patient had a previous history of total colectomy and an abdominal desmoid tumour within the context of Familial Adenomatous Polyposis (FAP), with a documented germline mutation in the Adenomatous Polyposis Coli (APC) gene. The eyelid lesion was biopsied and the histological analysis of the three small tissue fragments received revealed fragments with cutaneous–conjunctival lining displaying a subepithelial proliferation of basaloid nests with peripheral palisading, compatible with primitive hair follicles. There were images of anastomosis between different basaloid nests, which had their connection to the epithelial lining preserved. The stroma had high cellularity and sometimes primitive mesenchymal papillae were evident. Pleomorphism was absent, mitotic figures were barely identified, and no necrosis was seen. The basaloid nests did not have epithelial–stromal retraction nor mucin deposits. A diagnosis of BFH was proposed, which was later confirmed after surgical excision of the whole eyelid lesion. No evidence of carcinoma was present. This case illustrates the main features of the rare benign eyelid BFH. The standard medical or surgical approach of these lesions remains to be firmly established. Nearly nine months after surgical excision our patient remains well without signs of disease recurrence.
publishDate 2022
dc.date.none.fl_str_mv 2022-01-07
2022-01-07T00:00:00Z
dc.type.status.fl_str_mv info:eu-repo/semantics/publishedVersion
dc.type.driver.fl_str_mv info:eu-repo/semantics/article
format article
status_str publishedVersion
dc.identifier.uri.fl_str_mv https://hdl.handle.net/1822/78289
url https://hdl.handle.net/1822/78289
dc.language.iso.fl_str_mv eng
language eng
dc.relation.none.fl_str_mv Lamas, N.J.; Rodrigues, A.P.; Araújo, M.; Vizcaíno, J.R.; Coelho, A. Basaloid Follicular Hamartoma of the Eyelid: A Case Report and Literature Review about an Unusual Lesion in the Ocular Region. Diagnostics 2022, 12, 140. https://doi.org/10.3390/diagnostics12010140
2075-4418
10.3390/diagnostics12010140
140
https://www.mdpi.com/2075-4418/12/1/140
dc.rights.driver.fl_str_mv info:eu-repo/semantics/openAccess
eu_rights_str_mv openAccess
dc.format.none.fl_str_mv application/pdf
dc.publisher.none.fl_str_mv Multidisciplinary Digital Publishing Institute (MDPI)
publisher.none.fl_str_mv Multidisciplinary Digital Publishing Institute (MDPI)
dc.source.none.fl_str_mv reponame:Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)
instname:Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informação
instacron:RCAAP
instname_str Agência para a Sociedade do Conhecimento (UMIC) - FCT - Sociedade da Informação
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reponame_str Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)
collection Repositório Científico de Acesso Aberto de Portugal (Repositórios Cientìficos)
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