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Trichoadenoma of the upper eyelid: case report and literature review

Carlos Izquierdo-Rodriguez12, Emilio Dorronzoro-Ramirez3, Santiago Nieto-Llanos12
Ophthalmol J 2022;7:1-5.

Abstract

Background: Trichoadenoma of Nikolowski is a rare and benign tumor of the hair follicle. It was first described in 1958. The clinical appearance of trichoadenoma can be confused with basal cell carcinoma, and a differential diagnosis must be made with this entity and with other benign lesions such as epidermal cyst, seborrheic keratosis, actinic keratosis, and more exceptionally with comedo.

Case presentation: We report a case of a 45-year-old woman with a pigmented lesion in the left eyelid mimicking comedo. Histopathology study showed lesions containing keratinous cysts surrounded by lymphocytic components in the dermis and hair shaft among the cystic keratin, which is unusual in these tumors. The lesions were diagnosed as trichoadenoma.

Conclusion: We suggest that all excised eyelid lesions be sent to histopathological study.

Case report

DOI: 10.5603/OJ.2022.0001

Trichoadenoma of the upper eyelid: case report and literature review

Carlos Izquierdo-Rodriguez12Emilio Dorronzoro-Ramirez3Santiago Nieto-Llanos12
1Henares University Hospital, Spain
2Facultad de Ciencias de la Salud. Universidad Francisco de Vitoria, Pozuelo de Alarcón, Spain
3Sanitas La Moraleja Hospital, Madrid, Spain

Corresponding author:

Carlos Izquierdo-Rodriguez, M.D., Henares University Hospital, Spain, e-mail: drizqdo@gmail.com

This article is available in open access under Creative Common Attribution-Non-Commercial-No Derivatives 4.0 International (CC BY-NC-ND 4.0) license, allowing to download articles and share them with others as long as they credit the authors and the publisher, but without permission to change them in any way or use them commercially

ABSTRACT
Background: Trichoadenoma of Nikolowski is a rare and benign tumor of the hair follicle. It was first described in 1958. The clinical appearance of trichoadenoma can be confused with basal cell carcinoma, and a differential diagnosis must be made with this entity and with other benign lesions such as epidermal cyst, seborrheic keratosis, actinic keratosis, and more exceptionally with comedo.
Case presentation: We report a case of a 45-year-old woman with a pigmented lesion in the left eyelid mimicking comedo. Histopathology study showed lesions containing keratinous cysts surrounded by lymphocytic components in the dermis and hair shaft among the cystic keratin, which is unusual in these tumors. The lesions were diagnosed as trichoadenoma.
Conclusion: We suggest that all excised eyelid lesions be sent to histopathological study.
Key words: trichoadenoma; hair follicle tumor; adnexal tumor
Ophthalmol J 2022; Vol. 7, 1–5

Introduction

Trichoadenoma is a rare benign tumor derived from the infundibular part of the pilosebaceous unit. Nikolowski [1] described the first case in 1958 as “organoid folicular hamartoma”. Clinically, it appears as a solitary tumor. It involves adults without sex predilection [2] and is most commonly presented on the face (57.5%) and the buttocks (24.2%) and may involve eyelid, shoulder [2], lip [3], vulva [4], penis [5]. Only three cases reported are located on the eyelid, and the present case represents the fourth found in the eyelid reported in the literature.

The clinical appearance of trichoadenoma can be misleading and suggest a diagnosis of basal cell carcinoma, epidermal cyst, or seborrheic keratosis.

Histologically, it is characterized by numerous horny cysts lined with stratified squamous epithelium that resembles the external root lamia of the hair follicle.

The aim of this report is to review the literature and collect the cases reported to date about trichoadenoma and detail the clinical and histologic features of a trichoadenoma of the eyelid, a rare location of this lesion. Table 1 lists the published cases, ordered by year of publication, and the location of the lesion and the previous clinical diagnosis are also presented.

Table 1. Summary of reported cases of trichoadenoma

Author

Year

Age

Sex

Location

Clinical diagnosis

1

Nikolowski [1]

1958

63

M

Thigh

Sklerosierter nodulus cutaneus

29

Rahbari [11]

1978

41

M

Buttock

Cyst

33

F

Buttock

Cyst

38

F

Face (pretemporal)

Basal cell epithelioma

67

F

Face (suborbital)

Basal cell epithelioma

38

F

Face (jaw)

Basal cell epithelioma

31

M

Face (nose)

Basal cell epithelioma

46

F

Face (cheek)

Basal cell epithelioma

59

M

Face (retroauricular)

Basal cell epithelioma

1013

Nikolowski [12]

1978

24

F

Face (nasolabial)

Trichoepithelioma

21

M

Face (nose)

Basal cell epithelioma

24

F

Face (glabella)

Basal cell epithelioma

64

M

Face (angle of the eye)

Millium

14

Garcia e Silva

1982

50

M

Face (preauricular)

Atheroma

15

Undeutsch

1984

26

M

Face (parietal)

Not stated

16

Kanitakis

1986

32

F

Buttock

Histiocytofibroma/dermatofibrosarcoma

1718

Hey [13]

1987

66

M

Buttock

Atheroma

63

M

Face (suborbital)

Not stated

19

Bonvalet

1988

38

F

Neck

Atheroma

2021

Jaqueti [14]

1989

72

F

Face (suborbital)

Seborrheic keratosis

82

F

Face (cheek)

Seborrheic keratosis

22

Yazaki

1978

72

M

Upper arm

Not stated

23

Shishiba

1984

19

M

Face (rorehead)

Dermatofibroma

24

Iwasaki

1984

58

M

Buttock

Not stated

25

Hirokawa

1987

25

F

Face (forehead)

Solitary trichoepithelioma

26

Yamaguchi

1992

41

M

Buttock

Atheroma

27

Sieron [3]

1993

74

F

Upper lip

Not stated

28

Banuls

1995

50

M

Buttock

Not stated

29

Pavithran [5]

1996

M

Penis

Not stated

3036

Reibold [15]

1998

17

M

Face (forehead)

Basalioma

69

F

Buttock

Cyst

36

F

Back

Cyst

33

F

Face (suborbital)

Cyst

25

F

Face (forehead)

Nevus

7

F

Cheek

Cyst

45

M

Chin

Fibroma

37

Shields [6]

1998

80

F

Lower eyelid

Basal cell carcinoma

38

Yu

1998

61

F

Shoulder

Not stated

39

Kuwokawa [16]

2005

45

F

Check

Not stated

40

Gonzalez-Vela

2007

29

F

Face (forehead)

Melanocytic nevus

41

Lee W.S.

2007

5

M

Face (forehead)

Nevus sebaceous

42

Lee J.H.

2008

1

M

Nose

Not stated

43

Krishna

2008

28

M

Buttock

Fibroma

4462

Shimanovich [2]

2010

37

M

Face

58

M

Face

57

M

Face

25

M

Face

59

M

Face

13

M

Face

44

M

Face

23

F

Face

19

M

Face

33

F

Buttock

37

M

Buttock

37

M

Buttock

67

M

Buttock

40

M

Thigh

30

F

Thigh

56

F

Thigh

60

F

Shoulder

46

M

Shoulder

41

M

Breast

63

Matos

2011

76

F

External auditory canal

Not stated

64

Lever [7]

2012

63

M

Lower eyelid

Chalazion

65

Rashmi

2015

60

F

Vulva

Fibrosis lymphangioma circumscriptum

66

Arora

2013

28

F

Cheek

Lupus vulgar

67

Bombeccari

2015

44

M

Upper Lip

Not stated

68

Sangwaiya

2017

25

F

Vulva

Not stated

69

Cheng [8]

2019

54

M

Upper eyelid

Giant comedos

70

Pampena

2019

45

M

Eyebrow

Not stated

7182

Huet [10]

2020

12 F

5 M

Buttock 4

Thigh 1

Forehead 2

Cheek 3

Ear 1

Unknown 1

Not stated

83

Mir-Bonafe

2020

60

F

Chin

Millia cyst

Case report

A 45-year-old woman was referred to Ophthalmology Department for evaluation of a pigmented lesion on her left eyelid.

Four lesions in the inner third of the upper eyelid as a group of nodules stuffed with black material mimicking comedos (Fig. 1A) were founded on a slit-lamp exam.

01-Izquierdo-Rodriguez-1-html.jpg
Figure 1. A. Multiple nodular comedo-mimicking lesions on upper eyelid. B. Keratin cysts surrounded by mature squamous cells (H&E, ×4). C. White arrows shows hair shafts among cystic keratin, an unusual finding in these tumors (H&E, ×10). D. White arrow showing detailed hair shaft (H&E, x15)

The lesions were surgically removed under local anesthesia and sent for histopathological examination.

Histological exam revealed the existence of several cystic formations lined by squamous epithelium that resembled a follicular infundibulum. They were occupied by abundant keratotic material in which hair structures were recognized. In some cysts, there were projections from the epithelium towards the periphery that resembled abortive follicular bulbs and was surrounded by a lymphocytic inflammatory cyst at the dermal level (Fig. 1B).

Discussion

Trichoadenoma generally occurs as a solitary, nodular lesion that clinically resembles basal cell carcinoma, epidermal cysts, or seborrheic keratosis, and differential diagnosis of these lesions is needed. The lesions are usually isolated, asymptomatic, and slow-growing, with a size of less than 1.5 cm. The evolution of trichoadenoma is benign, and surgical excision is the preferred treatment.

This is the second reported case of comedo-like trichoadenoma on the upper eyelid to the best of the authors’ knowledge. There are only three reports of trichoadenoma affecting the eyelids [6–8].

The lesion reported by Shields et al. [6] was suspected to be a basal cell carcinoma affecting the lower eyelid. The lesion published by Lever was thought to be a sebaceous cell carcinoma that involved the lower eyelid [7], and Cheng reported a case mimicking comedo affecting the upper eyelid [8], as in the reported case.

Microscopic examination is necessary to make the diagnosis of trichoadenoma. The tumor is composed of keratin-filled cysts surrounded by mature squamous cells similar to the follicular infundibulum. Stromal fibroblastic inductions are evident around these cysts, sometimes associated with solid outgrowths of epithelium. The differential diagnosis with other cystic cutaneous neoplasms is based on the absence of basaloid cells (cystic basal cell carcinoma, seborrheic keratosis, or trichoepithelioma) and the absence of stromal induction in the case of epidermal cysts [9]. Interestingly, in our case, several hair shafts were identified among the cystic keratin, which is unusual in these tumors (Fig. 1CD).

Table 1 lists over 70 cases of trichoadenoma reported to date, and most of them are single cases. Shimanovich [2] in 2010 reported 19 cases, and Huet [10] in 2020 — 12 cases, and these are the largest case series. Another case series found in the literature was: Rahbari [11] in 1978 (8 cases), Nikolowski [12] in 1978 (4 cases), Hey [13] in 1987 (2 cases), Jaqueti [14] in 1989 (2 cases) and Reibold [15] in 1998 (7 cases). The histogenesis of trichoadenoma remains unclear. In terms of morphological differentiation, trichoadenoma is less mature than trichofolliculoma and more differentiated than trichoepithelioma. Trichoadenoma is characterized by a prominent cyst formation and resembles some features of trichoepithelioma. Both lesions retain Merkel cells as an element of differential diagnosis [2] regarding basal cell carcinoma. Regarding follicular neoplasms, trichofolliculoma is characterized by numerous secondary hair follicles radiating from primary central dilated hair follicles and a well-organized fibrovascular stroma. Immunohistochemical studies have shown cytokeratin (CK) 20 positive in trichoadenoma and trichoepithelioma as a specific marker for epithelial neuroendocrine cells of Merkel. In 2005 [16], Kurokawa reported a case with the immunohistochemical study of cytokeratins and found that CK10 and CK15 were present in trichoadenoma. CK10 may be found in most horn cysts with keratohyalin granules and CK15 in the outermost cells in horn cysts and the basaloid epithelium, suggesting that trichoadenoma differentiates towards the follicular infundibulum and follicular hair bulge of the outer root sheath.

Trichoadenoma is an underdiagnosed tumor, mainly due to its resemblance to common periocular lesions such as actinic keratosis, seborrheic keratoses, or epidermal cysts. These lesions are not routinely sent to the histopathological exam. Most of the histopathologically diagnosed cases of trichoadenoma are due to clinical suspicion of basal cell carcinoma and its referral to histopathological analysis.

Conclusions

Despite the studies focused on the immunohistochemical analysis of the lesions, the histopathological diagnosis continues to be the one that provides the most cost-benefit balance. We suggest referring all excised periocular lesions for a correct histopathological diagnosis, even those suspected of macroscopically benign or common lesions.

References

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