Basaloid follicular hamartoma of the eyelid in a pediatric patient

Jedrzynski, Nicole; Plum, William; Tran, Ann Q; Tooley, Andrea A.; Dowlatshahi, Mitra; Kazim, Michael

doi:10.1016/j.ajoc.2020.100855

Cited by 4 publications

(8 citation statements)

References 13 publications

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“…BFH is a rare benign malformative lesion involving the hair follicles, normally limited to the superficial dermis [ 1 ]. There are only three previous cases of BFH reported in the eyelid [ 4 , 5 , 6 ]. The first case was published in 2012, concerning an 86 year-old female patient that had an asymptomatic lesion in the left lateral lower eyelid evolving for approximately 2 years [ 4 ].…”

Section: Discussionmentioning

confidence: 99%

“…Afterwards, there was a report about a 52 year-old male patient, who presented with a 10 mm solitary, asymptomatic, hyperpigmented, slow-growing left upper eyelid tumour, increasing for 4 years [ 5 ]. The most recently reported case is relative to a paediatric diabetic female patient of 6 years old, who presented with a flesh coloured papule in the upper eyelid evolving for approximately two years [ 6 ]. Therefore, our case represents the fourth published case of eyelid BFH, illustrating the main features of this unusual benign entity, which is rarely described in the eyelid.…”

Section: Discussionmentioning

confidence: 99%

“…Clinically, BFH can present as a small individual skin-coloured to brown papule, or as multiple lesions located mainly in the face, scalp or trunk [ 1 ]. Interestingly, only three cases of BFH were previously reported in the eyelid [ 4 , 5 , 6 ]. Histologically, BFH is characterized by an epithelial proliferation of basaloid cells which can mimic benign hair follicle-based lesions or basal cell carcinoma (BCC), among other entities [ 1 ].…”

Section: Introductionmentioning

confidence: 99%

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Basaloid Follicular Hamartoma of the Eyelid: A Case Report and Literature Review about an Unusual Lesion in the Ocular Region

et al. 2022

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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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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Basaloid Follicular Hamartoma of the Eyelid: A Case Report and Literature Review about an Unusual Lesion in the Ocular Region

et al. 2022

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“…Although FMBFH generally presents at birth or in early childhood, milder variants may be recognized later in life [6,7]. Generalized forms of BFH are usually associated with systemic manifestations, which vary according to the subtype and can include cystic fibrosis, diffuse alopecia, and various autoimmune diseases (such as myasthenia gravis or systemic lupus erythematosus), [2,8,9].…”

Section: Case Discussionmentioning

confidence: 99%

Familial multiple basaloid follicular hamartoma

Cerejeira¹,

Gomes²,

Pacheco³

et al. 2021

DOJ

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“…“Kissing” lesions have, except in one case report, 2 only been reported with melanocytic nevi. To the best of our knowledge, there are only four reported cases of BFHs on the eyelid 3–6 . Here, we present an interesting case of “kissing” BFHs, and review the histopathologic and immunohistochemical characteristics.…”

Section: Introductionmentioning

confidence: 92%

“Kissing” basaloid follicular hamartomas of the eyelid

et al. 2022

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Basaloid follicular hamartomas (BFHs) are rare, benign, cutaneous adnexal tumors characterized by branching cords and anastomosing strands of basaloid cells in a loose, fibrous stroma. BFHs exhibit variable clinical presentations although they are commonly observed as skin-colored papules and are diagnosed based on histopathological features. Common systemic diseases associated with BFH include alopecia, myasthenia gravis, and palmoplantar pitting. BFH of the eyelid is extremely rare with only five cases reported in the literature to date. Congenital "kissing" lesions have only previously been reported with nevi. Here, we present a novel case of congenital "kissing" BFH of the right upper and right lower eyelid, and histopathological examination revealed intradermal nodules of basaloid cells forming reticulated strands, pseudohorn cysts, mucinous stroma, and palisading with CD34 and Bcl-2 expression in the stromal fibroblasts and periphery, respectively.

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Basaloid follicular hamartoma of the eyelid in a pediatric patient

Cited by 4 publications

References 13 publications

Basaloid Follicular Hamartoma of the Eyelid: A Case Report and Literature Review about an Unusual Lesion in the Ocular Region

Basaloid Follicular Hamartoma of the Eyelid: A Case Report and Literature Review about an Unusual Lesion in the Ocular Region

Familial multiple basaloid follicular hamartoma

“Kissing” basaloid follicular hamartomas of the eyelid

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