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E-IJD CASE REPORT
Year : 2015  |  Volume : 60  |  Issue : 2  |  Page : 214
Multiple trichofolliculomas mimicking multiple trichoepitheliomas


1 Department of Dermatology, Kasturba Medical College and Hospital, Manipal University, Manipal, Karnataka, India
2 Department of Pathology, Kasturba Medical College and Hospital, Manipal University, Manipal, Karnataka, India

Date of Web Publication3-Mar-2015

Correspondence Address:
Sudhir UK Nayak
Department of Dermatology, Kasturba Medical College and Hospital, Manipal University, Manipal - 576 104, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-5154.152591

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   Abstract 

Trichofolliculomas are benign hair follicle hamartomas which were initially considered as hair follicle tumors. Usually presenting as a solitary lesion associated with a tuft of vellus hairs, multiple trichofolliculomas are rare. Trichofolliculomas are characterized by a histopathological feature of dermal keratin cyst with cyst wall showing radiating hair follicles. We report this case for the rare presentation of multiple trichofolliculomas on the face which clinically mimicked multiple trichoepitheliomas.


Keywords: Mimic, multiple, trichoepitheliomas, trichofolliculomas


How to cite this article:
Nayak SU, Shenoi SD, Geetha V, Prabhu S, Nagel B. Multiple trichofolliculomas mimicking multiple trichoepitheliomas. Indian J Dermatol 2015;60:214

How to cite this URL:
Nayak SU, Shenoi SD, Geetha V, Prabhu S, Nagel B. Multiple trichofolliculomas mimicking multiple trichoepitheliomas. Indian J Dermatol [serial online] 2015 [cited 2020 Apr 8];60:214. Available from: http://www.e-ijd.org/text.asp?2015/60/2/214/152591

What was known?
Trichofolliculoma is usually solitary and multiple is rare. The characteristic histopathology is of a dermal keratin cyst with secondary follicles.



   Introduction Top


Trichofolliculoma although classified under hair follicle tumors, essentially are benign hamartomas of the hair follicle. [1] They are rare and usually present as solitary lesions on the face often with a tiny tuft of fine white hairs arising from the surface. [2] Majority of the case reports are of solitary type. We report a case of multiple trichofolliculomas that clinically resembled multiple trichoepitheliomas.


   Case Report Top


A 21-year-old male presented with multiple asymptomatic skin colored raised lesions on the face of 13 years duration. The lesions had gradually increased in size and number. There was a history of similar lesions in mother and sister. Clinical examination revealed multiple non-tender skin colored papules on the forehead, cheeks, nose, naso-labial folds and chin [Figure 1]. A clinical diagnosis of trichoepithelioma was considered and a biopsy done.
Figure 1: Multiple trichofolliculomas on the face

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Haematoxylin and eosin stained sections showed a dermal keratin cyst lined by squamous epithelium. Strands of secondary hair follicles were seen radiating from the wall of the cyst. The adjacent dermis showed pilosebaceous units and a partially ruptured hair follicle eliciting a giant cell response [Figure 2] and [Figure 3]. On the basis of the histopathological features, a final diagnosis of trichofolliculomas was made.
Figure 2: Dermal keratin cyst with secondary hair follicles (H and E, ×5)

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Figure 3: Secondary hair follicle from keratin cyst wall (H and E, ×20)

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   Discussion Top


Trichofolliculoma first described by Miescher. [3] is a rare hair follicle tumor that manifests as a solitary skin colored papule or nodule often with a central pore. [2] The characteristic feature is the tuft of fine white hair that projects from the pore. [2] Solitary trichofolliculomas occur primarily over the face, scalp and neck. Multiple trichofolliculomas are rare and have been reported on the upper extremities, [4] scrotum and penis, [5] and a patient with only two trichofolliculomas on ears with generalized multiple trichogenic myxomas. [6]

Trichofolliculoma usually appears in middle age. Childhood onset is rare. A patient with congenital trichofolliculoma has been described. [3]

The presence of vellus hairs in a tuft like pattern arising from the lesion is considered to a distinctive feature of trichofolliculoma. [7] However, trichofolliculomas undergo evolution during the course and thus, manifests with varying clinical and histopathological morphologies. [2],[4] The three stages of trichofolliculomas include early, full developed and late stages. [4]

Another useful aid to the diagnosis of trichofolliculoma is dermascopy showing peripheral radial dark brown projections resembling a crown and described as 'firework pattern'. These projections represent the nests of cells radiating from the follicular epithelium. [8]

The characteristic histopathological features of trichofolliculoma include a squamous epithelium lined keratin containing dermal cyst with radiating hair follicles from the cyst wall. These secondary hair follicles are interconnected with each other by epithelial strands in all cases. [2] These histopathological features tend to differentiate trichofolliculomas from other hair follicular tumors. Cohesive and branching clusters of keratinized squamous cells mixed with sebaceous cells are seen in fine needle aspiration cytology of trichofolliculoma. [9]

Two variants of trichofolliculomas include sebaceous trichofolliculoma and folliculosebaceous cystic hamartoma that may represent an evolutionary stage. [2],[7] Even though trichofolliculomas are considered to be benign, perineural invasion has been described in a patient as well as recurrence following excision. [10] Association of vulval trichoepitheliomas with vulvar intraepithelial neoplasia III has also been reported. [11]

Our patient presented with multiple skin colored papules resembling trichoepitheliomas and it was only the biopsy which confirmed the diagnosis of trichofolliculoma. In view of the long standing duration of the lesions, the classical picture of vellus hairs protruding from the nodule was not seen. Our case was very similar to the previously reported case of multiple trichofolliculomas with numerous 0.3 to 1.5 cm sized papules and nodules without any vellus hairs. [4]

Our initial diagnosis was multiple trichoepitheliomas in view of the childhood onset; positive family history and clustering of lesions in the nasolabial folds but the characteristic histological features such as horn cysts and basaloid epithelial islands in the dermis were absent.

We report this case for its rarity of occurrence, onset in childhood, presence of family history of similar lesions, the clinical resemblance to trichoepitheliomas and the absence of clinically demonstrable vellus hairs.

 
   References Top

1.
McCalmont TH. Adnexal Neoplasms. In: Bolognia JL, Jorizzo JL, Schaffer JV, editors. Dermatology. 3 rd ed. China: Elsevier Saunders; 2012. p. 1829-30.  Back to cited text no. 1
    
2.
Ahmed TS, Del Priore J, Seykora JT. Tumors of the epidermal appendages. In: Elder DE, Elenitsas R, Jaworsky C, Johnson Jr B, editors. Lever's Histopathology of the Skin. 10 th ed (South Asian). New Delhi: Wolters Kluwer (India); 2009. p. 854-9.  Back to cited text no. 2
    
3.
Sodeify M, Aghaei S. Trichofolliculoma on scalp. Indian J Deramtol 2004;49:79-80.  Back to cited text no. 3
    
4.
Choi CM, Lew BL, Sim WY. Multiple trichofolliculomas on unusual sites: A case report and review of the literature. Int J Dermatol 2013;52:87-9.  Back to cited text no. 4
    
5.
Nomura M, Hata S. Sebaceous trichofolliculoma on scrotum and penis. Dermatologica 1990;181:68-70.  Back to cited text no. 5
    
6.
Cohen C, Davis TS. Multiple trichogenic adnexal tumors. Am J Dermatopathol 1986;8:241-6.  Back to cited text no. 6
    
7.
Ghosh SK, Bandyopadhyay D, Barma KD. Perifollicular nodule on the face of a young man. Indian J Dermatol Venereol Leprol 2011;77:531-3.  Back to cited text no. 7
[PUBMED]  Medknow Journal  
8.
Panasiti V, Roberti V, Lieto P, Visconti B, Calvieri S, Perrella E. The "firework" pattern in dermoscopy. Int J Dermatol 2013;52:1158-9.  Back to cited text no. 8
    
9.
Alexander MP, Makhija P, Jayaseelan E. Fine needle aspiration cytology of trichofolliculoma: A case report. Indian J Pathol Microbiol 2007;50:870-2.  Back to cited text no. 9
[PUBMED]    
10.
Stern JB, Stout DA. Trichofolliculoma showing perineural invasion. Trichofolliculocarcinoma Arch Dermatol 1979;115:1003-4.  Back to cited text no. 10
    
11.
Peterdy GA, Huettner PC, Rajaram V, Lind AC. Trichofolliculoma of the vulva associated with vulvar intraepithelial neoplasia: Report of three cases and review of the literature. Int J Gynecol Pathol 2002;21:224-30.  Back to cited text no. 11
    

What is new?
Clinical resemblances to trichoepitheliomas. Childhood onset and family history of similar lesions.


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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    Abstract
   Introduction
   Case Report
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    References
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