|Year : 2015 | Volume
| Issue : 3 | Page : 780-782
Pilomatricoma with florid osseous metaplasia: A rare case report
Tushar Kambale, Banyameen Iqbal MBBS, MD , Atul Jain, Komal Sawaimul
Department of Pathology, Dr. D. Y. Patil Medical College, Hospital and Research Center, Pune, Maharashtra, India
|Date of Submission||13-May-2014|
|Date of Acceptance||18-Jan-2015|
|Date of Web Publication||22-Oct-2015|
Department of Pathology, Dr. D. Y. Patil Medical College Hospital and Research Centre, Pimpri, 411018 Pune, Maharashtra
Source of Support: None, Conflict of Interest: None
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|| |
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 . 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 . In 1922, Dubreuilh and Cazenave  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 , thus avoiding the word epithelioma, which carries the connotation of malignancy.
| Case report|| |
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.
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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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|| |
The most frequent anatomical location of pilomatricoma is the head and neck region, followed by the upper extremities ,. Most of the cases present as single nodules, but multiple occurrences have also been reported ,. Familial cases have been observed in association with disorders such as Gardner syndrome, Steinert disease and sarcoidosis . 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 ,. 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 . In a series of 27 cases studied by Zaman et al. , 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 ,.
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 . 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 . 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 . 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 .
The patient is on follow-up, and the follow-up period has been uneventful so far.
| Conclusion|| |
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|| |
Conflicts of interest
| References|| |
Alsaad KO, Obaidat NA, Ghazarian D. Skin adnexal neoplasm part 1: an approach to tumours of the pilosebaceous unit. J Clin Pathol 2007; 60
Malherbe A, Chenantais J. Note on calcified epithelioma of sebaceous glands. Prog Med 1880; 8
Dubreuilh W, Cazenave E. The calcified epithelioma: histological study. Ann Dermatol Syphilol 1922; 3
R, Helwig EB. Pilomatrixoma (calcifying epithelioma). Arch Dermatol 1961; 83
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
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
Kania M, Kaminska-Winciorek G, Krauze E, Brzezinska-Wcisto L, Kajor M. Multifocal type of pilomatrixoma. Adv Med Sci 2007; 52
SS, Gadre SA, Adegboyega P, Gadre AK. Multiple pilomatricomas: a case report and literature review. Ear Nose Throat J 2008; 87
N, Azmy A, Carachi R, Raine PA, Macfariane JH, Howatson AG. Pilomatrixoma: accuracy of clinical diagnosis. J Pediatr Surg 2006; 41
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.
Zaman S, Majeed S, Rehman F. Pilomatricoma - study on 27 cases and review of literature. Biomedica 2009; 25
Dutta R, Boadle R, Ng T. Pilomatrix carcinoma: case report and review of the literature. Pathology 2001; 33
Mikhaeel NG, Spittle MF. Malignant pilomatrixoma with multiple local recurrences and distant metastases: a case report and review of the literature. Clin Oncol2001; 13
Pirouzmanesh A, Reinisch JF, Gonzalez-Gomez I, Smith EM, Meara JG. Pilomatrixoma: a review of 346 cases. Plast Reconstr Surg 2003; 112
Graham JL, Merwin CF. The tent sign of pilomatricoma. Cutis 1978; 22
Wagner AM. A 12-year-old girl with an enlarging nontender nodule on the arm. Pediatr Ann 2006; 35
Agarwal RP, Handler SD, Matthews MR, Carpentieri D. Pilomatrixoma of the head and neck in children. Otolaryngol Head and Neck Surg 2001; 125
[Figure 1], [Figure 2], [Figure 3]