Indian Journal of Dermatology
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CASE REPORT
Year : 2009  |  Volume : 54  |  Issue : 5  |  Page : 16-18
Sebaceous carcinoma of cheek arising in a lesion of solar keratosis


1 Department of Pathology, Madras Medical College, Chennai - 600 003, India
2 Department of Dermatology, Madras Medical College, Chennai - 600 003, India

Correspondence Address:
Ponnuswamy Karkuzhali
No. 12/3, Main Road, Karpagavinayagar colony, Alapakkam, Chennai, Tamilnadu -600116
India
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   Abstract 

Cutaneous extraocular sebaceous carcinoma is a rare tumor that frequently occurs on face and scalp. Sebaceous carcinoma is not considered to be associated with ultra violet radiation exposure. We describe a case of sebaceous carcinoma of the right side of the cheek occurring in a lesion of solar keratosis in a 44-year-old female, who is a farmer by occupation. Actinic keratosis carries about 8-20% risk for malignant transformation into squamous cell carcinoma. Other malignant lesions that have been reported to occur in actinic keratosis include basal cell carcinoma, sebaceous carcinoma and trichilemmal carcinoma. To our knowledge, this is the third case to be reported in English language literature. Unlike the two cases reported previously, this has occurred in a younger patient and presented as an ulcerative, invasive lesion of 3cm size. The association of actinic keratosis and sebaceous carcinoma indicate that solar UVR is probably of importance in the pathogenesis of sebaceous carcinoma, as in the case of squamous cell carcinoma.


Keywords: Actinic keratosis, cutaneous lesion, extraocular type, head and neck region, sebaceous carcinoma, solar keratosis


How to cite this article:
Karkuzhali P, Gomathy N, Ahamed PB. Sebaceous carcinoma of cheek arising in a lesion of solar keratosis. Indian J Dermatol 2009;54:16-8

How to cite this URL:
Karkuzhali P, Gomathy N, Ahamed PB. Sebaceous carcinoma of cheek arising in a lesion of solar keratosis. Indian J Dermatol [serial online] 2009 [cited 2014 Apr 24];54:16-8. Available from: http://www.e-ijd.org/text.asp?2009/54/5/16/45434



   Introduction Top


Actinic keratoses are premalignant epithelial lesions occurring on chronically light-exposed adult skin that carry a low risk of progression to squamous cell carcinomas. Other rare malignancies reported include sebaceous carcinoma [1] and trichilemmal carcinoma. [2] Only two cases of sebaceous carcinoma in association with actinic keratosis have been reported in the English language literature so far. One case was an intraepidermal carcinoma and another case presented initially as an intraepidermal lesion that became invasive one-year later. [1] In this context, it should be noted that intraepidermal sebaceous carcinoma is difficult to distinguish clinically and histologically from actinic keratosis.


   Case History Top


A 44-year-old, light colored female with brown eyes, a farmer by occupation, was admitted to the Department of Dermatology, Madras Medical College, Chennai, for the complaints of an ulcer over the right cheek of eight months duration and pigmented macules, erythematous patches and plaques of four years duration. The lesion started initially as a papule four years ago and was excised. It recurred at the same site one year later and got eroded and evolved into an ulcer. She also gave history of increased wrinkles, freckles and lentigenes over sun-exposed areas since 16years ofage.

Dermatological examination revealed a single, tender ulcer of about 32cm size with necrotic base. The edges were raised and the ulcer was found fixed to the underlying structures [Figure 1]. Other findings included a nodule of 2x1 cms size over the right forearm and multiple, discrete, flat to elevated erythematous scaly and non-scaly patches, macules and plaques of varying sizes over solar damaged skin. Mucosa, hair, nails, palms and soles were found to be normal. Clinical examination also revealed bilateral, enlarged cervical lymph nodes about four in number that varied in size from 0.5-1cm. A provisional clinical diagnosis of OCA type IIA with actinic keratosis and malignant transformation was made. Biopsies were taken from ulcer the over the right cheek and the nodule over the forearm. Histopathological diagnosis of the ulcer was sebaceous carcinoma and the nodule was actinic keratosis. Fine needle aspiration of the biopsy of the cervical lymph node showed features of chronic non-specific lymphadenitis. Ultrasonography of the abdomen and pelvis and colonoscopy to search for associated lesions did not reveal any positive findings. Subsequent to histological diagnosis, surgical excision with wide margins and skin grafting was done at the department of Plastic surgery, Madras Medical College, Chennai.

Pathology

The excised mass measured 4x3x1cm with an ulcer about 32cm in the center. Cut section revealed a linear, solid, and gray-white mass just beneath the ulcer.

Microscopic examination revealed an ulcerated squamous epithelium with hypertrophic type of actinic keratotic changes and features of intraepidermal sebaceous carcinoma [Figure 2], covering an unencapsulated neoplasm in the dermis composed of lobules of varying sizes. Lobules showed larger oval cells of varying sizes with clear or pale, bubbly cytoplasm. Nuclei were centrally located, showed nuclear grooves, clear chromatin, and prominent nucleoli. They also showed moderate degree of atypia and occasional mitotic figures [Figure 3]. Focal areas showed squamoid differentiation of tumor cells with necrosis.

Tumor showed infiltration into adjacent salivary gland tissue [Figure 4]. A periparotid Lymphnode showed secondary carcinomatous deposit. Microscopic examination of the forearm nodule showed features of bowenoid type of actinic keratosis.


   Discussion Top


Extra ocular sebaceous carcinoma constitutes of all cases of sebaceous carcinomas [3] that has a potential for aggressive behavior. Extraocular sebaceous carcinomas commonly occur in the head and neck region. Males are twice more commonly affected than females and patients are usually in the seventh decade. Rare cases in children have been reported. [4] It has been reported in association with eczema, actinic keratosis, xeroderma pigmentosum, multiple sclerosis, [5] nevus sebaceous, [6] Bowen's disease and Muir-Torre syndrome.

Frozen sections reveal lipid droplets in the cytoplasm of cells and immunoreactivity for keratin, cytokeratin, EMA, Leu-M1, anti breast carcinoma associated antigen-225 (CU18), anti CA 15.3 [7] and Thomson-Friedenreich antigen. [8] A histological grading system for sebaceous carcinomas exists, [9] but the prognostic significance of this is not yet well established. Histologically, the sebaceous carcinomas have to be distinguished from basal cell carcinoma with sebaceous differentiation, clear cell squamous cell carcinoma, clear cell hidradenocarcinoma and balloon cell malignant melanoma.

Poor prognostic factors include multicentricity, size>1cm in diameter, poor differentiation, extensive tissue infiltration, vascular or lymphatic involvement, [3] pagetoid change, duration of more than six months and previous irradiation. Mortality rate varies from 20-22%. Wide excision is the treatment of choice for well-differentiated lesions. In cases of poorly differentiated lesions, wide excision is followed by radiotherapy and regular follow-up of skin, lymph nodes and other organ systems.

This case is a rare example of sebaceous carcinoma occurring in a lesion of solar keratosis and is probably the third case to be reported in the literature in such category. An analysis of p53 and PCNA expression in cases of actinic keratosis has revealed lower positivity in atrophic and acantholytic type with higher activity in hypertrophic and bowenoid type. [10] The occurrence of sebaceous carcinomas in actinic keratosis indicates a possible role for UVR in the etiopathogenesis of extraocular type of sebaceous carcinomas.

 
   References Top

1.Ansai S, Mihara I. sebaceous carcinoma arising on actinic keratosis. Eur J Dermatol 2000;10:385-8.  Back to cited text no. 1  [PUBMED]  [FULLTEXT]
2.Misago N, Tanaka T, Kohda H. Trichilemmal carcinoma occurring in a lesion of solar keratosis. J Dermatol 1993;20:358-64.  Back to cited text no. 2  [PUBMED]  
3.Chapter 33. Tumours of cutaneous appendages. In : WeedonD, Strutton G, editors. Skin pathology. Churchill Livingstone: Elsevier Sciences Ltd; 2002. p. 876-7.  Back to cited text no. 3    
4.Omura NE, Collison DW, Perry AE, Myers LM. Sebaceous carcinoma in children. J Am Acad Dermatol 2002;47:950-3.  Back to cited text no. 4  [PUBMED]  [FULLTEXT]
5.Akhtar S, Ozak K, Roulier RG. Multiple sebaceous adenomas and extra ocular. Sebaceous carcinoma with multiple sclerosis: Case report and review of literature. J Cutan Med Surg 2001;5:490-5.  Back to cited text no. 5    
6.de Giorgi V, Massi D, Brunasso G, Mannone F, Soyer HP, CarliP. Sebaceous carcinoma arising from nevus sebaceus: Case report. Dermatol Surg 2003;29:105-7.  Back to cited text no. 6    
7.Ansai S, Mitsuhashi Y, Kondo S, Manabe M. Immunohistochemical differentiation of extra ocular sebaceous carcinoma from other skin cancers. J Dermatol 2004:31:998-1008.  Back to cited text no. 7    
8.Hassanein AM, Al-Quran SZ, Kantor GR, Pauporte M, TelangGH, Spielvogel RL. Thomson-Friedenreich (T) antigen: A possible tool for differentiating sebaceous carcinoma from its simulators. Appl immunohistochem mol morphol 2001;9:250-4.  Back to cited text no. 8    
9.Rao NA, Hidayat AA, McLean IW, Zimmerman LE. Sebaceous carcinomas of the ocular adnexa: A clinicopathologic study of 104 cases with five-year follow-up. Hum Pathol 1982;13:113-22.  Back to cited text no. 9  [PUBMED]  
10.Ramzi ST, Maruno M, Khaskhely NM, Khan MA, TakamiyagiA, Uezato H, et al . Bowen's disease: p53 and PCNA expression pattern correlate with the number of desmosomes. J Dermatol 2002;29:562-72.  Back to cited text no. 10    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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    Abstract
    Introduction
    Case History
    Discussion
    References
    Article Figures

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