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 Table of Contents  
LETTER TO EDITOR
Year : 2015  |  Volume : 2  |  Issue : 2  |  Page : 43-45

Solitary stony hard plaque over the scalp in a young adult: A case report


1 Department of Dermatology, Adichunchanagiri Institute of Medical Sciences, Mandya, Karnataka, India
2 Consultant Dermatologist, Nirmal Skin and Hair Clinic, Bangalore, Karnataka, India

Date of Web Publication6-Jan-2016

Correspondence Address:
Mukunda Ranga Swaroop
Department of Dermatology, Adichunchanagiri Institute of Medical Sciences, Mandya, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2349-6029.173417

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How to cite this article:
Swaroop MR, Vasudevan P, Nischal KC, Sathyanarayana BD. Solitary stony hard plaque over the scalp in a young adult: A case report. Indian J Dermatopathol Diagn Dermatol 2015;2:43-5

How to cite this URL:
Swaroop MR, Vasudevan P, Nischal KC, Sathyanarayana BD. Solitary stony hard plaque over the scalp in a young adult: A case report. Indian J Dermatopathol Diagn Dermatol [serial online] 2015 [cited 2021 May 10];2:43-5. Available from: https://www.ijdpdd.com/text.asp?2015/2/2/43/173417

Sir,

Osteoma cutis (OC) is a rare and benign disease characterized by the formation of bone tissue in dermis and subcutaneous tissue. It presents in children and adults with a female preponderance. [1] It is classified as primary OC (15%) wherein bone tissue formation occurs in skin without any pre-existing lesions or is associated with syndromes such as Albright's hereditary osteodystrophy (AHO) and secondary OC (85%) wherein osseous tissue formation occurs in preexisting lesions. [2] To date, very few cases of isolated plate-like OC have been reported worldwide. We are describing a case of adult-onset plate-like OC over the scalp of 4 years' duration.

A 28-year-old man presented with a slowly growing, asymptomatic, raised lesion over the scalp for the past 4 years. There was no history of itching, pain or bleeding at the site of the lesion. He had no history of prior trauma or any previous cutaneous lesions on the scalp. There was no history of similar lesions in the family members. On clinical examination, a solitary, well-defined, stony hard plaque of size 2 cm × 1 cm, was present over the vertex of the scalp [Figure 1]. He did not have any features suggestive of AHO.
Figure 1: A solitary, well-defined stony hard plaque of size 2 × 1 cm over the vertex


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Complete blood count (CBC), serum calcium and phosphorus, serum electrolytes, and liver and renal function tests were within normal limits. Punch biopsy from the lesion revealed a focus of ossification in the dermis [Figure 2]. The ossified focus showed hollow areas containing capillaries, ground substance, collagen, and fibroblasts. There was a foreign body granuloma around the ossified focus, composed of foreign body giant cells, epithelioid cells, histiocytes, neutrophils, and lymphocytes [Figure 3]. The adjoining epidermis showed hypergranulosis and basket weave orthohyperkeratosis.
Figure 2: H and E stain (100×): Focus of ossification in the dermis showing hollow areas containing capillaries, ground substance, collagen, and fibroblasts


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Figure 3: H and E stain (100×): Foreign body granuloma around the ossified focus, composed of foreign-body giant cells, epithelioid cells, histiocytes, neutrophils, and lymphocytes


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Based on history, clinical examination, and histopathological findings, adult-onset plate-like OC was diagnosed.

OC is a rare and benign disease, characterized by the formation of mature lamellar bone within the dermis and subcutaneous tissue. The first case of OC was reported in 1858 by Wilckets. [3] It is classified into primary and secondary forms. [4] Primary OC may occur either de novo or in association with syndromes such as AHO, fibrodysplasia ossificans progressiva or progressive osseous heteroplasia (POH). [5],[6]

Secondary OC is the most common variant (85%), wherein the ossification process occurs in pre-existing inflammatory or neoplastic skin lesions such as acne vulgaris, pilomatricoma, trauma, venous stasis, syringoma, epidermoid cysts, scleroderma, scars, and basal cell carcinoma. [7] In our case, there was no clinical and histopathological evidence of a pre-existing skin lesion.

The pathogenesis of primary OC is not known. Burgdorf and Nasemann proposed two theories. The first theory suggested anomalous migration of osteoblasts to the skin, whereas the second theory stated metaplasia of fibroblast to osteoblast in the dermis due to mutation in the gene GNAS1, which encodes for a protein known to regulate adenyl cyclase activity. [8]

OC presents in adults and children with a greater female preponderance. Clinically, primary OC manifests in four clinical patterns:

  1. Solitary osteom,a presenting as an asymptomatic nodule, occurring at any site, commonly in adults;
  2. Widespread OC, presenting as multiple generalized subcutaneous nodules in the neonatal period;
  3. Multiple miliary osteomas of the face, presenting in women with/without acne; and
  4. Congenital plate-like osteoma in neonates or infants, which manifests as asymptomatic large plaque on the scalp or the extremities. [9]


However, our patient presented with an asymptomatic stony hard plaque over the scalp of only 4 years' duration.

Primary OC may also be associated with syndromes such as, AHO, which is characterized clinically by short stature, round facies, brachydactyly, developmental delay, café au lait macules, and fibrous dysplasia of long bones. It is also associated with hypocalcemia, hyperphosphatemia, and elevated serum parathyroid hormone levels, [10],[11] which in our case was excluded by physical and laboratory evaluation.

Worret and Burgdorf first coined the term "congenital plate-like osteoma cutis" in 1978 with the following diagnostic criteria:

  1. Lesion present at birth or within the first year of life;
  2. No evidence of abnormal calcium or phosphorous metabolism;
  3. No history of infection, trauma, or other predisposing events; and
  4. Presence of at least one bony plate with or without cutaneous osteomas. [12]


Since that time, there have been other reports of plate-like OC that do not present in the first few years of life. Till date, less than 15 cases of acquired, idiopathic, plate- or plaque-like primary OC have been reported, [13],[14] the most common location being the head and scalp in men. [15] Our patient presented with a solitary asymptomatic plaque on the scalp of 4 years' duration with no secondary findings both clinically and histopathologically. Hence we arrived at the diagnosis of acquired, adult-onset plate-like OC.

Histologically, OC is composed of a well-circumscribed nodule of mature lamellar bone, completely calcified and inhabited by osteocytes, often containing marrow spaces, present within the dermis. [16]

Surgical excision is the treatment of choice for plate-like OC. Other modalities of treatment are erbium:YAG laser, dermabrasion, and topical application of retinoids. [17],[18]

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Ayaviri NA, Nahas FX, Barbosa MV, Farah AB, de Arimatéia Mendes J, Ferreira LM. Isolated primary osteoma cutis of the head: Case report. Can J Plast Surg 2006;14:33-6.  Back to cited text no. 1
    
2.
Cottoni F, Dell′ Orbo C, Quacci D, Tedde G. Primary osteoma cutis. Clinical, morphological, and ultrastructural study. Am J Dermtopathol 1993;15:77-81.  Back to cited text no. 2
    
3.
Wilhelmsen HR, Bereston ES. Treatment of osteoma cutis. Cutis 1984;33:481, 483.  Back to cited text no. 3
    
4.
Boschert MT, Puckett CL. Osteoma cutis of the hand. Plast Reconstr Surg 2000;105:1017-8.  Back to cited text no. 4
    
5.
Kaplan FS, Shore EM. Progressive osseous heteroplasia. J Bone Miner Res 2000;15:2084-94.  Back to cited text no. 5
    
6.
Talsania N, Jolliffe V, O′Toole EA, Cerio R. Platelike osteoma cutis. J Am Acad Dermatol 2011;64:613-5.  Back to cited text no. 6
    
7.
Aguinaga F, Trope B, Piñeiro-Maceira J, Ramos-E-Silva M. Miliary osteoma cutis: A case report. Case Rep Dermatol Med 2014;2014:347829.  Back to cited text no. 7
    
8.
Burgdorf W, Nasemann T. Cutaneous osteomas: A clinical and histopathologic review. Arch Dermatol Res 1977;260:121-35.  Back to cited text no. 8
    
9.
Sanmartín O, Alegre V, Martinez-Aparicio A, Botella-Estrada R, Aliaga A. Congenital platelike osteoma cutis: Case report and review of the literature. Pediatr Dermatol 1993;10:182-6.  Back to cited text no. 9
    
10.
Jeong KH, Lew BL, Sim WY. Osteoma cutis as the presenting feature of albright hereditary osteodystrophy associated with pseudopseudohypoparathyroidism. Ann Dermatol 2009;21:154-8.  Back to cited text no. 10
    
11.
Kucukemre Aydin B, Yazganoglu KD, Baykal C, Buyukbabani N, Ucar A, Bas F, et al. Osteoma cutis. Pediatr Int 2013;55:257-8.  Back to cited text no. 11
    
12.
Douri T, Shawaf AZ. Plate-like cutaneous osteoma on the scalp. Dermatol Online J 2006;12:17.  Back to cited text no. 12
    
13.
Haro R, Revelles JM, Angulo J, Fariña Mdel C, Martín L, Requena L. Plaque-like osteoma cutis with transepidermal elimination. J Cutan Pathol 2009;36:591-3.  Back to cited text no. 13
    
14.
Vashi N, Chu J, Patel R. Acquired plate-like osteoma cutis. Dermatol Online J 2011;17:1.  Back to cited text no. 14
    
15.
Aneiros-Fernandez J, Husein-ElAhmed H, Orgaz-Molina J, O′Valle Ravassa F, Arias-Santiago S. A man with infiltrated and indurated plaque on the forehead. Dermatol Online J 2010;16:12.  Back to cited text no. 15
    
16.
Calonje E, Brenn T, Lazar A, Mckee PH. McKee′s Pathology of the Skin. 4 th ed. USA: Elsevier; 2012. p. 1751.  Back to cited text no. 16
    
17.
Ugur K, Sevgi K, Nihat P, Hulya U, Sezgin Z, Gulgun K, et al. Osteoma cutis: Report of one case. J Clin Exp Dermatol Res 2011;2:135.  Back to cited text no. 17
    
18.
Wu M, Wang Y, Zhang D, Jia G, Bu W, Fang F, et al. A case of giant primary osteoma cutis successfully treated with tissue expansion and surgical excision. Indian J Dermatol Venereol Leprol 2011;77:79-81.  Back to cited text no. 18
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