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CASE REPORT
Year : 2020  |  Volume : 12  |  Issue : 3  |  Page : 132-134  

Nevus lipomatosus cutaneous superficialis associated with trichofolliculoma: An uncommon duet of rare pathologies


1 Department of Pathology, Nazareth Hospital, Shillong, Meghalaya, India
2 Department of Surgery, Nazareth Hospital, Shillong, Meghalaya, India

Date of Submission19-Feb-2020
Date of Decision21-Mar-2020
Date of Acceptance08-Apr-2020
Date of Web Publication14-Aug-2020

Correspondence Address:
Dr. Harveen Kaur Gulati
Department of Pathology, Nazareth Hospital, Laitumkhrah, Shillong - 793 003, Meghalaya
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijt.ijt_21_20

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   Abstract 


Nevus lipomatosus cutaneous superficialis (NLCS) and trichofolliculoma (TF) are both rare cutaneous hamartomatous lesions. However, the coexistence of NLCS and TF as a single lesion has been reported only in two other cases, that too in middle-aged individuals. We report the first case of NLCS associated with TF in pediatric age group and also, to the best of our knowledge, the first case reported from India.

Keywords: Nevus lipomatosus cutaneous superficialis, pediatric age group, trichofolliculoma


How to cite this article:
Agarwal A, Gulati HK, Das JK. Nevus lipomatosus cutaneous superficialis associated with trichofolliculoma: An uncommon duet of rare pathologies. Int J Trichol 2020;12:132-4

How to cite this URL:
Agarwal A, Gulati HK, Das JK. Nevus lipomatosus cutaneous superficialis associated with trichofolliculoma: An uncommon duet of rare pathologies. Int J Trichol [serial online] 2020 [cited 2020 Sep 21];12:132-4. Available from: http://www.ijtrichology.com/text.asp?2020/12/3/132/292097




   Introduction Top


Nevus lipomatosus cutaneous superficialis (NLCS) and trichofolliculoma (TF) are both rare cutaneous hamartomatous lesions.[1],[2] However, the coexistence of NLCS and TF as a single lesion has been reported only in two other cases that too in middle-aged individuals.[3],[4] To the best of our knowledge, we report the first case of NLCS associated with TF in the pediatric age group and also the first case reported from India.


   Case Report Top


Our case was a 7-year-old female with a cauliflower-like growth on the left forearm, which was noticed when it suddenly increased in size following bamboo stick injury. The child was brought to our hospital and a small growth was noted. Given the history, a provisional diagnosis of foreign body granuloma was made. On excision, a small sinus draining thick white material was noted and was sent to the histopathology department. We received a single skin covered tissue bit measuring 2.5 cm × 1.5 cm × 0.8 cm along with a central cauliflower growth measuring 2.0 cm × 1.0 cm × 0.8 cm. Cut surface of the cauliflower-like lesion was yellowish and communicating with the tissue underlying the skin [Figure 1].
Figure 1: Upper part of the image showing cut surface of the specimen with protruding yellow lesion extending into underlying tissue on one of the shoulders. Lower part of the image showing an image of the section on the slide which highlights the above features (H and E)

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On microscopic examination, the polypoid lesion was overlined by the skin with underlying tissue showing adipocytes admixed with fibrous tissue. In addition, multiple, thin strands of the squamous epithelium were seen arising from the overlying epidermis. Also noted was a large cystically dilated hair follicle-like structure with abundant keratin material within the lumen [Figure 2]. The surrounding area showed strands of the squamous epithelium with branching as well as numerous horn cysts and sebaceous glands associated with it. The surrounding tissue showed numerous ruptured horn cysts associated with foreign body giant cell reaction. A diagnosis of “NLCS associated with TF” was made.
Figure 2: A photomicrograph showing a large cystically dilated hair follicle-like structure with abundant keratin material within the lumen. From the epithelial lining are seen strands of squamous epithelium associated with numerous horn cysts and sebaceous glands (H and E, ×40)

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   Discussion Top


NLCS is a rare hamartomatous lesion which presents as flesh-colored or yellow-colored papules or nodules, at birth or within the first two decades of life, and is most commonly seen in the gluteal region and rarely in the scalp and face.[1] Similarly, TF is also a rare cutaneous hamartoma presenting in adulthood as a solitary nodule in head-and-neck region with central umblication and tuft of the hair.[2],[3] The occurrence of NLCS associated with other lesions has been reported, but rarely, which include abortive hair germ-like structures, hyperplastic pilosebaceous units, fibrofolliculomas, folliculosebaceous cystic hamartoma (FSCH), and cylindroma.[5],[6],[7],[8],[9],[10]

However, the coexistence of NLCS and TF as a single lesion has been reported only in two other cases that too in middle-aged individuals.[3],[4] Both these cases presented in the late age group but had been presented for several years and presented on the forearm and lower leg. Our case presented in this pediatric age group probably because of the bamboo stick injury which brought the lesion to attention. Our case presented on the forearm which appears to be the preferred site (limbs). Histologically, both the cases showed similar histopathologic features like ours. Even though some authors believe FSCH and TF to be a part of the same continuum, the presence of abortive hair follicles in TF appears to be an important feature which distinguishes it from FSCH.[11]

From the above cases and the present case, an observation can be made that NLCS associated with TF develops in childhood, has no sex predilection, presents in sites where NLCS or TF is extremely rare, and has predilection for the limbs. Further, this rare entity may show variable gross features, due to which it can often be misdiagnosed. Even though NLCS is not known to recur, TF often shows recurrence. Malignant transformation is not known to occur in either of the lesions. Hence, excision is indicated with follow-up.

We also believe that NLCS is highly under-reported, given its benign nature, and is not as rare as it is believed to be. Therefore, it can be concluded that variants and lesions associated with NLCS exist, which have variable clinical presentation and histopathologic features and should be looked for while diagnosing a NLCS with atypical clinical features.

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.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Weedon D. Weedon's Skin Pathology E-Book: Expert Consult-Online and Print. Elsevier Health Sciences; 2009. p. 846.  Back to cited text no. 1
    
2.
Bharti JN, Dey B, Gautam P, Desai P, Kamal V. Trichofolliculoma presenting as lobulated mass: A rare presentation. Int J Trichology 2014;6:19-20.  Back to cited text no. 2
    
3.
Bancalari E, Martínez-Sánchez D, Tardío JC. Nevus lipomatosus superficialis with a folliculosebaceous component: Report of two cases. Patholog Res Int 2011;2011:105973.  Back to cited text no. 3
    
4.
Sakanoue M, Kawai K, Kanekura T. Nevus lipomatosus cutaneous superficialis associated with trichofolliculoma on the lower leg. Indian J Dermatol 2013;58:330.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Jones EW, Marks R, Pongsehirun D. Naevus superficialis lipomatosus. A clinicopathological report of twenty cases. Br J Dermatol 1975;93:121-33.  Back to cited text no. 5
    
6.
Inoue M, Ueda K, Hashimoto T. Nevus lipomatosus cutaneus superficialis with follicular papules and hypertrophic pilo-sebaceous units. Int J Dermatol 2002;41:241-3.  Back to cited text no. 6
    
7.
Yun SJ, Kim EJ, Kim SJ, Lee SC, Won YH, Lee JB. The association of naevus lipomatosus with pilosebaceous abnormalities including fibrofolliculoma. Br J Dermatol 2005;153:209-10.  Back to cited text no. 7
    
8.
Brasanac D, Boricic I. Giant nevus lipomatosus superficialis with multiple folliculosebaceous cystic hamartomas and dermoid cysts. J Eur Acad Dermatol Venereol 2005;19:84-6.  Back to cited text no. 8
    
9.
Kang H, Kim SE, Park K, Son SJ, Song KY. Nevus lipomatosus cutaneous superficialis with folliculosebaceous cystic hamartoma. J Am Acad Dermatol 2007;56:S55-7.  Back to cited text no. 9
    
10.
Yu R, Alowami S. Cylindroma with stromal adipose tissue metaplasia versus arising in a background of nevus lipomatosus. Case Rep Pathol 2014;2014:203298.  Back to cited text no. 10
    
11.
Pierard GE, Lapiere CM. Nevi of connective tissue. A reappraisal of their classification. Am J Dermatopathol 1985;7:325-33.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2]



 

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