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CASE REPORT
Year : 2015  |  Volume : 28  |  Issue : 3  |  Page : 780-782

Pilomatricoma with florid osseous metaplasia: A rare case report


Department of Pathology, Dr. D. Y. Patil Medical College, Hospital and Research Center, Pune, Maharashtra, India

Date of Submission13-May-2014
Date of Acceptance18-Jan-2015
Date of Web Publication22-Oct-2015

Correspondence Address:
Banyameen Iqbal
Department of Pathology, Dr. D. Y. Patil Medical College Hospital and Research Centre, Pimpri, 411018 Pune, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1110-2098.165825

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  Abstract 

Pilomatricoma, formerly known as Pilomatrixoma is a benign dermal and/or subcutaneous tumour. It most commonly affects children and adolescents; however, a second smaller peak of onset is also reported in the elderly. It is slightly more common in females. This tumour was first described by Malherbeand in 1880 as a benign, subcutaneous tumor arising from hair cortex cells. The name pilomatrixoma was proposed by Forbis and Helwig in 1961, thus avoiding the word epithelioma, which carries the connotation of malignancy. Although osseous metaplasia in pilomatrixoma is a very rare occurrence but there are a few cases reported in the literature which has shown focal or florid osseous metaplasia. We are hereby presenting a case of pilomatrixoma with florid osseous mataplasia.

Keywords: Benign adnexal tumours, osseus metaplasia, pilomatrixoma


How to cite this article:
Kambale T, Iqbal B, Jain A, Sawaimul K. Pilomatricoma with florid osseous metaplasia: A rare case report. Menoufia Med J 2015;28:780-2

How to cite this URL:
Kambale T, Iqbal B, Jain A, Sawaimul K. Pilomatricoma with florid osseous metaplasia: A rare case report. Menoufia Med J [serial online] 2015 [cited 2020 Apr 4];28:780-2. Available from: http://www.mmj.eg.net/text.asp?2015/28/3/780/165825


  Introduction Top


Pilomatricoma, formerly known as pilomatrixoma, is a benign dermal and/or subcutaneous tumour. It most commonly affects children and adolescents; however, a second smaller peak of onset is also reported in the elderly. It is slightly more common in women [1]. This tumour was first described by Malherbe and Chenantais in 1880 as a benign, subcutaneous tumor arising from hair cortex cells. Since then, this uncommon entity has been called calcifying epithelioma of Malherbe [2]. In 1922, Dubreuilh and Cazenave [3] described the characteristic histological features: islands of epithelial cells, including shadow cells and giant cells. The name pilomatrixoma was proposed by Forbis and Helwig in 1961 [4], thus avoiding the word epithelioma, which carries the connotation of malignancy.


  Case report Top


A 52-year-old female patient presented to the Dermatology Department with complaints of painless chest-wall swelling for the last 18 years. The swelling measured 4 × 2 × 2 cm, was firm to hard in consistency, was nontender and the overlying skin was normal. The swelling was freely mobile and not attached to any underlying structures. All baseline investigations were carried out and were within normal limits. A clinical diagnosis of sebaceous cyst was made. The swelling was excised and sent for histopathological examination.

Gross examination

A skin-covered tissue specimen measuring 3 × 2 × 2 cm, yellowish in colour and hard in consistency was received in the Pathology Department. The specimen was subjected to decalcification before processing [Figure 1].
Figure 1: Gross specimen showing yellow-coloured, well-circumscribed tumour underlying the skin.

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Histopathological examination

Multiple sections studied showed a benign tumour underlying the epidermis. The tumour consisted of a biphasic pattern of keratinized ghost cells surrounded by a variable number of basaloid cells along with areas of ossification and multiple osteoclastic giant cells. The keratinized ghost cells had central unstained areas representing lost nuclei and usually had distinct cell borders. Some focal areas of calcification were also seen. These features thus favoured pilomatricoma with florid osseous metaplasia [Figure 2] and [Figure 3].
Figure 2: Photomicrograph shows squamous epithelium lined tissue with underlying florid osseous metaplasia (HE ×20).

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Figure 3: Photomicrograph showing ghost cells and foci of osseous metaplasia (HE ×100).

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  Discussion Top


The most frequent anatomical location of pilomatricoma is the head and neck region, followed by the upper extremities [5],[6]. Most of the cases present as single nodules, but multiple occurrences have also been reported [7],[8]. Familial cases have been observed in association with disorders such as Gardner syndrome, Steinert disease and sarcoidosis [5]. A few cases of malignant pilomatricoma metastasizing to the lung, bone, brain, abdominal organs, skin, and lymph nodes have also been described in the literature [8],[9]. Although osseous metaplasia in pilomatrixoma is a very rare occurrence, there are a few cases reported in the literature that have shown osseous metaplasia, focal or florid [10]. In a series of 27 cases studied by Zaman et al. [11], they found only one case of pilomatrixoma with osseous differentiation. A rare malignant counterpart, pilomatrix carcinoma, has been described, and ~90 cases have been reported in the literature. It is locally aggressive and can recur. In several cases, it has demonstrated metastases. Many key features are similar between these benign and malignant counterparts; the primary differentiating characteristics include a high mitotic rate with atypical mitoses, central necrosis, infiltration of the skin and soft tissue and invasion of blood and lymphatic vessels [12],[13].

On presentation, as in this case, palpation of a superficial firm nodule that is not painful or tender is characteristic; however, 32% of patients in a series of 346 cases presented with pain and tenderness [14]. Most commonly, the overlying skin is of normal colour and texture; however, the examiner may observe the tent sign, consisting of flattening of some portion or the entire surface of the tumour with angulation resembling the side of a tent, often seen only by stretching the skin [15]. This has been attributed to attachment of the tumour to the overlying epidermis; the associated bluish or reddish discolouration is due to the growth of blood vessels into the overlying skin [16]. Although pilomatrixomas are usually solitary, multiple lesions have been reported in association with genetic disorders, such as myotonic dystrophy, Gardner syndrome, xeroderma pigmentosum and basal cell nevus syndrome [17].

The patient is on follow-up, and the follow-up period has been uneventful so far.


  Conclusion Top


The present case highlights the importance of considering pilomatrixoma and its various differentiations, especially osseous differentiation, in the clinical and pathologic differential diagnosis of dermal or subcutaneous nodule even in locations other than the head and neck region. This is true especially for pathologists, as they play a pivotal role in the diagnosis of these lesions. Pathologists should keep in mind the variability of the these lesions in terms of its various lines of differentiation, especially osseous.


  Acknowledgements Top


Conflicts of interest

None declared.

 
  References Top

1.
Alsaad KO, Obaidat NA, Ghazarian D. Skin adnexal neoplasm part 1: an approach to tumours of the pilosebaceous unit. J Clin Pathol 2007; 60 :129-144.  Back to cited text no. 1
    
2.
Malherbe A, Chenantais J. Note on calcified epithelioma of sebaceous glands. Prog Med 1880; 8 :826-837.  Back to cited text no. 2
    
3.
Dubreuilh W, Cazenave E. The calcified epithelioma: histological study. Ann Dermatol Syphilol 1922; 3 :257-268.  Back to cited text no. 3
    
4.
R, Helwig EB. Pilomatrixoma (calcifying epithelioma). Arch Dermatol 1961; 83 :606-617.  Back to cited text no. 4
[PUBMED]    
5.
S, Nicollas R, Roman S, Magalon G, Triglia JM. Pilomatrixoma of the head and neck in children; a study of 38 cases and a review of the literature. Arch Otolaryngol Head Neck Surg 1998; 124 :1239-1242.  Back to cited text no. 5
[PUBMED]    
6.
MY, Lan MC, Ho CY, Li WY, Lin CZ. Pilomatrixoma of the head and neck in children: a retrospective review of 179 cases. Arch Otolaryngol Head Neck Surg 2003; 129 :1327-1330.  Back to cited text no. 6
    
7.
Kania M, Kaminska-Winciorek G, Krauze E, Brzezinska-Wcisto L, Kajor M. Multifocal type of pilomatrixoma. Adv Med Sci 2007; 52 :251-252.  Back to cited text no. 7
    
8.
SS, Gadre SA, Adegboyega P, Gadre AK. Multiple pilomatricomas: a case report and literature review. Ear Nose Throat J 2008; 87 :230-233.  Back to cited text no. 8
    
9.
N, Azmy A, Carachi R, Raine PA, Macfariane JH, Howatson AG. Pilomatrixoma: accuracy of clinical diagnosis. J Pediatr Surg 2006; 41 :1755-1758.  Back to cited text no. 9
    
10.
Kawakami M, Yoshimura K, Ito K, Tanaka H, Higashikawa M, Hyo S. Two cases of pilomatrixoma in the cheek. Bull Osaka Med Coll 2004; 50:13-17.  Back to cited text no. 10
    
11.
Zaman S, Majeed S, Rehman F. Pilomatricoma - study on 27 cases and review of literature. Biomedica 2009; 25 :69-72.  Back to cited text no. 11
    
12.
Dutta R, Boadle R, Ng T. Pilomatrix carcinoma: case report and review of the literature. Pathology 2001; 33 :248-251.  Back to cited text no. 12
    
13.
Mikhaeel NG, Spittle MF. Malignant pilomatrixoma with multiple local recurrences and distant metastases: a case report and review of the literature. Clin Oncol2001; 13 :386-389.  Back to cited text no. 13
    
14.
Pirouzmanesh A, Reinisch JF, Gonzalez-Gomez I, Smith EM, Meara JG. Pilomatrixoma: a review of 346 cases. Plast Reconstr Surg 2003; 112 :1784-1789.  Back to cited text no. 14
    
15.
Graham JL, Merwin CF. The tent sign of pilomatricoma. Cutis 1978; 22 :577-580.  Back to cited text no. 15
[PUBMED]    
16.
Wagner AM. A 12-year-old girl with an enlarging nontender nodule on the arm. Pediatr Ann 2006; 35 :445-447.  Back to cited text no. 16
    
17.
Agarwal RP, Handler SD, Matthews MR, Carpentieri D. Pilomatrixoma of the head and neck in children. Otolaryngol Head and Neck Surg 2001; 125 :510-515.  Back to cited text no. 17
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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