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Year : 2021  |  Volume : 64  |  Issue : 1  |  Page : 195-196
“Solitary fibrofolliculoma of the eyelid: A less known entity”

1 Ophthalmic Pathology Laboratory, LV Prasad Eye Institute, Hyderabad, Telangana, India
2 Operation Eyesight Universal Institute for Eye Cancer, L V Prasad Eye Institute, Hyderabad, Telangana, India

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Date of Submission24-Jan-2020
Date of Decision11-Mar-2020
Date of Acceptance08-Jun-2020
Date of Web Publication8-Jan-2021

How to cite this article:
Mishra DK, Jakati S, Kaliki S. “Solitary fibrofolliculoma of the eyelid: A less known entity”. Indian J Pathol Microbiol 2021;64:195-6

How to cite this URL:
Mishra DK, Jakati S, Kaliki S. “Solitary fibrofolliculoma of the eyelid: A less known entity”. Indian J Pathol Microbiol [serial online] 2021 [cited 2022 Jan 19];64:195-6. Available from: https://www.ijpmonline.org/text.asp?2021/64/1/195/306525

Fibrofolliculomas are benign hamartomatous tumors characterized by the proliferation of perifollicular mesenchyme and follicular components. Histological exams revealed that the lesions entities lie somewhere between perifollicular fibroma and trichofolliculoma.[1]

Solitary lesions though are extremely rare, only five case reports were available worldwide (Medline search) and only two such reports are of an eyelid tumor.[2],[3],[4],[5],[6] Herein, we report a solitary fibrofolliculoma diagnosed in the right lower eyelid, exhibiting classical histological picture in an otherwise-healthy 50-year-old man.

A 50-year-old male presented with a swelling in his right lower lid for the past 6 months, which was gradually increasing in size. On examination, a 3 cm × 3cm × 2 cm firm, mobile, and painless swelling involving lateral two-thirds of the lower lid was noticed [Figure 1]a. The patient's visual acuity was 20/60 in the right eye andthe left eye 20/50 without aid. The anterior and posterior segments including fundus examination of the right and left eye were quite unremarkable except cataract in the left eye. There were no significant systemic anomalies and lymphadenopathy found.
Figure 1: (a) Clinical photograph of the right eye showing a polypoidal lesion in the lower eyelid. (b) Gross photograph showing solid and cystic area. Microphotograph of fibofolliculoma showing, (c) A well-defined tumor with prominent pilosebaceous unit and keratin filled cyst in the stroma, (H and E stain, 4×). (d) The proliferation of perifollicular fibrous sheath with myxoid changes surrounding the infundibular portion of the dilated hair follicles, (H and E stain, 10×). (e) Histopathological features of fibrofolliculoma, (H and E stain, 0.7×, Digital scanner view)

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Based on these findings clinically diagnosed as sebaceous gland carcinoma, the patient subsequently underwent excision biopsy and histopathological examination.

The postoperative period was uneventful and at the last follow-up period, 3 months later, there was no evidence of any recurrence and systemic examination did not reveal any syndromic association.

Gross pathological examination revealed a skin covered with firm polypoidal grayish-white mass measuring 18 mm × 13 mm × 8 mm with the cut surface being cystic and contained thick mucoid material [Figure 1]b. Microscopic examination showed a well-defined tumor with prominent pilosebaceous unit and keratin filled cyst in the stroma [Figure 1]c. There was the proliferation of perifollicular fibrous sheath with myxoid changes surrounding the infundibular portion of the dilated hair follicles [Figure 1]d. Multiple epithelial strands were seen arising from the infundibulum which was anastomosing with each other and with follicles. Multiple large cysts were seen in the dermis, few have a surface opening, and the cyst was lined by cuboidal to multilayered squamous cells with apical snouts and lumen was filled with lamellated keratinous material. Histopathological examination confirmed the presence of fibrofolliculoma [Figure 1]e.

Fibrofolliculoma was first described as a part of Birt, Hogg, and Dubé (BHD), an autosomal dominant syndrome characterized by multiple fibrofolliculomas, pulmonary cysts, pneumothorax, renal cysts, and renal cell carcinoma. Nearly 15 cases have been reported since then and these reported cases had multiple lesion and presented as trichodiscoma, acrochordons, and connective tissue nevus.[7] Wang et al. (2020) reported a solitary fiborofolliculoma in a 68-year-old female who presented with an asymptomatic mass on the right upper eyelid which was not associated with BHD.[6] Our case is similar to the reported case in which we also not got a syndromic association. Solitary fibrofolliculomas are sporadic and do not follow an inheritance pattern. Clinically, fibrofolliculoma is asymptomatic and presents with 2–4 mm lesions that are yellow-white to skin-colored, smooth, and dome-shaped. Generally, the age of onset for solitary fibrofolliculoma is the sixth decade, whereas the multiple hereditary forms have an early onset, and usually they present in the third decade of life.[4] In the spectrum of benign follicular neoplasms, fibrofolliculoma appears to be intermediate between perifollicular fibroma (purely of mesodermal origin) and trichofolliculoma (purely of epithelial origin). This creates some difficulty in determining a fibrofolliculoma, which is sometimes thought to be indistinguishable from these tumors. However, a fibrofolliculoma frequently has visible central hair, keratotic plug, dell, or umbilication, whereas trichofolliculoma has features of telangiectasia and peripheral location of hair that may help distinguish it from fibrofolliculoma.[1] Microscopically, fibrofolliculoma shows a well-circumscribed dermal tumor with a central well-formed dilated hair follicle that sometimes contains keratin material. The neoplastic epithelial cells proliferate from the central hair follicles and form thin anastomosing cords.

To conclude, solitary fibrofolliculoma is a rare tumor of the eyelid and on simple ophthalmic examination, it is not possible to diagnose asymptomatic cases. Solitary fibrofolliculoma should be considered in firm nodular lesions of the eyelid and histopathological examination is mandatory for the confirmation of diagnosis. Although recurrences are rare, all such patients should be kept on close follow-up.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

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Conflicts of interest

There are no conflicts of interest.

   References Top

Weintraub R, Pinkus H. Multiple fibrofolliculomas (Birt-Hogg-Dubé) associated with a large connective tissue nevus. J Cutan Pathol 1977;4:289-99.  Back to cited text no. 1
Scully K, Bargman H, Assaad D. Solitary fibrofolliculoma. J Am Acad 1984;11:361-3.  Back to cited text no. 2
Mo HJ, Park CJ, Yi JY. A case of solitary fibrofolliculoma. Korean J Dermatol 2001;39:602-4.  Back to cited text no. 3
Kim KH, Choi JH, Lee YS. A case of solitary fibrofolliculoma. Korean J Dermatol 1984;22:672-4.  Back to cited text no. 4
Bhattacharyya S, Bhattacharyya S, Maitra A. Solitary fibrofolliculoma of the eyelid. Curr Indian Eye Res 2015;2:83-5.  Back to cited text no. 5
Wang W, Cheng J. Solitary fibrofolliculoma of the upper eyelid in a 68-year old female: A case report. BMC Ophthalmol 2020;20:97.  Back to cited text no. 6
Birt AR, Hogg GR, Dubé WJ. Hereditary multiple fibrofolliculomas with trichodiscomas and acrochordons. Arch Dermatol 1977;?113:1674-7.  Back to cited text no. 7

Correspondence Address:
Dilip K Mishra
Department of Ophthalmic Pathology Laboratory, LV Prasad Eye Institute, Road No.02, Banjara Hills, KAR Campus, Hyderabad - 500 034, Telangana
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/IJPM.IJPM_57_20

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