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CASE REPORT
Year : 2018  |  Volume : 19  |  Issue : 1  |  Page : 65-67

Recurrent pilomatricoma of the thigh: An unusual site of presentation


Department of Dermatology, Venereology and Leprology, Regional Institute of Medical Sciences, Imphal, Manipur, India

Date of Web Publication28-Dec-2017

Correspondence Address:
Vanlalhriatpuii Hmar
Department of Dermatology, Venereology and Leprology, Regional Institute of Medical Sciences, Imphal - 795 001, Manipur
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpd.IJPD_72_16

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  Abstract 

Pilomatricoma is a benign tumor of the hair matrix. It is also known as calcifying epithelioma of Malherbe and is commonly located in head, neck, and upper extremities. A 10-year-old female patient presented with a solitary skin-colored firm nodule on her left mid-thigh which progressed to 1.5 cm diameter within 4 months. Surgical excision had been conducted 1 year ago at the same site. Clinical examination revealed a single, firm, well-circumscribed, non-tender, freely mobile nodule with irregular surface and hyperpigmented borders including surrounding skin. Histopathological examination showed dermis displaying benign adnexal tumor tissue with sheets of ghost cells, clusters of basaloid cells, and extensive areas of dystrophic calcification. Histopathological confirmation prior excision should be ideally performed. Although the tumor is most common in head and neck region, clinicians should be aware that tumors can arise rarely in the lower limbs as well.

Keywords: Calcifying epithelioma of Malherbe, lower limb, pilomatricoma, unusual site


How to cite this article:
Hmar V, Thokchom N, Kshetrimayum S. Recurrent pilomatricoma of the thigh: An unusual site of presentation. Indian J Paediatr Dermatol 2018;19:65-7

How to cite this URL:
Hmar V, Thokchom N, Kshetrimayum S. Recurrent pilomatricoma of the thigh: An unusual site of presentation. Indian J Paediatr Dermatol [serial online] 2018 [cited 2019 Aug 22];19:65-7. Available from: http://www.ijpd.in/text.asp?2018/19/1/65/206081


  Introduction Top


Pilomatricoma is a benign tumor considered to be a hamartoma of the hair matrix composed of cells resembling those of the hair matrix, cortex, and inner root sheath. It is also known as calcifying epithelioma of Malherbe. It may occur at any age but is frequently seen in children. The lesion is usually solitary, deep dermal, or subcutaneous tumor 3–30 mm diameter in size. Most commonly, they are located in head, neck, and upper extremities. Malignant change is very rare. Surgical excision is the treatment of choice. However, recurrences can occur due to incomplete excision.[1]


  Case Report Top


A 10-year-old female patient with an unremarkable medical history presented to us with a solitary skin-colored firm papule with purulent discharge on her left mid-thigh which had gradually progressed in size over 4 months into a firm nodule. Surgical excision had been conducted 1 year ago without any histopathological confirmation for similar lesion at the same site. Clinical examination revealed a single, firm, well-circumscribed, nontender, freely mobile nodule with irregular surface and hyperpigmented borders including surrounding skin on the medial surface of left mid-thigh [Figure 1]. The nodule measured approximately 1.5 cm diameter on the surface [Figure 2]. Inguinal lymphadenopathy was absent. Histopathological examination showed dermis displaying benign adnexal tumor tissue with sheets of ghost cells, clusters of basaloid cells, and extensive areas of dystrophic calcification. Few multinucleated giant cells with lymphocytic infiltration in the stroma were also noted [Figure 3].
Figure 1: Single indurated nodule on left inner thigh

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Figure 2: Close up image of tumor measuring approximately 1.5 cm in diameter

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Figure 3: Histopathology of tumor with dermis displaying benign adnexal tumor tissue, sheets of ghost cells, clusters of basaloid cells, and extensive areas of dystrophic calcification. Few multinucleated giant cells with lymphocytic infiltration in the stroma

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  Discussion Top


Pilomatricoma is a relatively uncommon skin tumor which can occur at any age but has increased frequency of occurrence in pediatric age as well as in the sixth decade.[2]

As the condition is uncommon, clinical diagnosis is usually non specific and requires histopathological confirmation prior surgical excision. Other investigative procedures that can be conducted before excision include fine-needle aspiration cytology (FNAC), ultrasonography, X-ray, computerized tomography (CT) scan and magnetic resonance imaging. FNAC with Giemsa stain may show basaloid cells, shadow (ghost) cells, calcification, nucleated squamous cells, and giant cells.[3],[4],[5] FNAC, however, may have drawbacks as it can lead to misdiagnosis if it is used as the sole investigative technique.[6] X-rays and CT scans may show fine or spotted calcifications and isointensity compared to skeletal muscles. Ultrasonography may show well-defined, oval to round, heterogeneous hypo or hyperechoic subcutaneous mass with hypoechoeic rim representing connective tissue capsule. Hyperechoic areas if present correspond to calcification.[1],[7],[8],[9] FNAC, X-ray, and ultrasonography were not performed in our case.

Most excised tumors are diagnosed as unspecified lumps with differential diagnoses ranging from sebaceous cyst, tuberculoma, hemangioma, keloid, cervical lymph node, etc.[10]

In most studies conducted, the tumors were usually solitary with size <2 cm in diameter.[10],[11] Our patient presented with a solitary tumor of 1.5 cm diameter size.

The most common site of presentation for pilomatricoma is head and neck area, followed by upper limb, trunk, and lower limb in decreasing order of frequency.[7],[10],[11],[12],[13],[14],[15] Our patient presented with a single firm to hard nodule in the left inner thigh. Tumors occurring in the lower limbs are unusual, and their clinical diagnosis, therefore, requires high degree of suspicion.[16]

Complete surgical excision is the mainstay of treatment. However, recurrences can and do occur after excision.[11],[13] Our patient had undergone surgical excision 1 year ago and reported to us with similar complaint at the same site. Malignant transformation is uncommon. Rarely, pilomatrical carcinoma may result from conventional pilomatricoma.[2]

Pilomatricoma, due to its uncommon nature of the occurrence, requires clinical suspicion and histopathological confirmation for appropriate diagnosis. Since recurrences can occur after surgical excision and can be alarming for both the clinician and patient, histopathological confirmation prior excision should be conducted to confirm benign nature of the disease. Although the tumor is most common in head and neck region, clinicians should be aware that tumors can arise rarely in the lower limbs as well.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Calonje E. Tumours of skin appendages. In: Burns T, Breathnach S, Cox N, Griffiths C, editors. Rook's Textbook of Dermatology. 8th ed. West Sussex (UK): Wiley-Blackwell; 2010. p. 53.12.  Back to cited text no. 1
    
2.
McCalmont T. Adnexal neoplasm. In: Bolognia JL, Jorizzo JL, Schaffer JV, editors. Dermatology. 3rd ed. Spain: Elsevier Limited; 2012. p. 1835.  Back to cited text no. 2
    
3.
Gupta R, Verma S, Bansal P, Mohta A. Pilomatrixoma of the arm: A rare case with cytologic diagnosis. Case Rep Dermatol Med 2012;2012:257405.  Back to cited text no. 3
    
4.
Khandeparkar SG, Joshi AR, Ail D, Kothikar V, Kulkarni MM. Giant pilomatricoma of the arm: A diagnostic challenge for cytopathologist. Int J Trichology 2016;8:43-5.  Back to cited text no. 4
    
5.
Sinhasan SP, Jadhav CR, Bhat RV, Amaranathan A. Pilomatrixoma – Presented as hypopigmented tender nodule: Diagnosed by FNAC: A case report with review of literature. Indian J Dermatol 2013;58:405.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Gupta M, Gupta V, Kumar R, Jhajj K. Pilomatrixoma: A diagnostic pitfall in fine needle aspiration cytology. JEMDS 2014;3:1691-7.  Back to cited text no. 6
    
7.
Kashima K, Igawa K, Takamori K, Yoshioka I, Sakoda S. A case of pilomatrixoma in the cheek in a 7-year-old girl. Oral Sci Int 2012;9:26-8.  Back to cited text no. 7
    
8.
Hwang JY, Lee SW, Lee SM. The common ultrasonographic features of pilomatricoma. J Ultrasound Med 2005;24:1397-402.  Back to cited text no. 8
    
9.
Garioni E, Danesino GM, Madonia L. Pilomatricoma: Sonographic features. J Ultrasound 2008;11:76-8.  Back to cited text no. 9
    
10.
Zaman S, Majeed S, Rehman F. Pilomatricoma – Study on 27 cases and review of literatures. Biomedica 2009;25:69-72.  Back to cited text no. 10
    
11.
Fonseca R, Filho J, Araujo I, Filho A, Pereira N, Carvalho E, et al. Pilomatricoma: Calcifying epithelioma of Malherbe. Rev Bras Cir Plást 2012;27:605-10.  Back to cited text no. 11
    
12.
Ieni A, Todaro P, Bonanno AM, Catalano F, Catalano A, Tuccari G. Limits of fine-needle aspiration cytology in diagnosing pilomatrixoma: A series of 25 cases with clinico-pathologic correlations. Indian J Dermatol 2012;57:152-5.  Back to cited text no. 12
[PUBMED]  [Full text]  
13.
Guinot-Moya R, Valmaseda-Castellon E, Berini-Aytes L, Gay-Escoda C. Pilomatrixoma. Review of 205 cases. Med Oral Patol Oral Cir Bucal 2011;16:e552-5.  Back to cited text no. 13
    
14.
Agrawal R, Kumar P. Pilomatricoma-unveiling the ghost story: Report of 5 cases with review of the literature. Int J Case Rep Images 2015;6:193-7.  Back to cited text no. 14
    
15.
Garg LN, Arora S, Gupta S, Gupta S, Singh P. Pilomatricoma: Forget me not. Indian Dermatol Online J 2011;2:75-7.  Back to cited text no. 15
[PUBMED]  [Full text]  
16.
Zaidi N, Tandon N, Lal N, Verma R. Pilomatricoma at uncommon site: A case report. Int J Curr Med Appl Sci 2014;5:20-2.  Back to cited text no. 16
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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