Indian Journal of Dermatology
  Publication of IADVL, WB
  Official organ of AADV
Indexed with Science Citation Index (E) , Web of Science and PubMed
 
Users online: 4016  
Home About  Editorial Board  Current Issue Archives Online Early Coming Soon Guidelines Subscriptions  e-Alerts    Login  
    Small font sizeDefault font sizeIncrease font size Print this page Email this page


 
Table of Contents 
CASE REPORT
Year : 2011  |  Volume : 56  |  Issue : 4  |  Page : 435-438
A solitary crateriform ulcer of the lower lip: A case report with review of literature


Department of Oral Medicine and Radiology, I.T.S. Centre for Dental Studies and Research, Muradnagar, Ghaziabad 201 206 (U.P.), India

Date of Web Publication10-Sep-2011

Correspondence Address:
Sunil Chaudhary
House No. 166, Sector 14, Vasundhara, Ghaziabad 201 206, (U.P.)
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-5154.84755

Rights and Permissions

   Abstract 

Keratoacanthoma (KA) is a relatively common skin lesion. We report a case of KA-mimicking squamous cell carcinoma in a 40-year old smoker, who was also having speckeled leukoplakia on the buccal mucosa. The present case highlights the importance of histopathological diagnosis, as the treatment in latter case is aggressive, which is unnecessary if attempted with KA. Moreover, the present report provides an overview of such lesions, which could be encountered in clinical situations for which definitive diagnosis is of paramount importance before the starting of treatment plan.


Keywords: Keratoacanthoma, psudo carcinomatous, Sq cell carcinoma, Keratcarcinoma


How to cite this article:
Chauhan A, Chaudhary S, Agnihotri P G, Aadithya B. A solitary crateriform ulcer of the lower lip: A case report with review of literature. Indian J Dermatol 2011;56:435-8

How to cite this URL:
Chauhan A, Chaudhary S, Agnihotri P G, Aadithya B. A solitary crateriform ulcer of the lower lip: A case report with review of literature. Indian J Dermatol [serial online] 2011 [cited 2019 Jun 16];56:435-8. Available from: http://www.e-ijd.org/text.asp?2011/56/4/435/84755



   Introduction Top


Keratoacanthoma (KA) is a distinctive benign tumor that displays rapid growth with clinical and histologic pattern often suggestive of squamous cell carcinoma. [1] It may be considered as "an abortive malignancy which rarely progresses into an invasive squamous cell carcinoma." [2] The term KA was first used by Rook and Whimster (1950) to describe a common, self-healing epithelioma of the skin occurring mainly on the exposed areas of the elderly people as an infiltrating, self-limited, localized growth of squamous cells. [3],[4] These lesions occur as painless, rapidly growing, raised nodules of size 1-2 cm, exhibiting a centrally depressed, keratin-filled crater descriptively referred to as "volcano.". [5],[6] The peak incidence of KA is between the ages of 45 and 69 years with a majority of tumors occurring on exposed parts of the skin, lips, cheeks, nose, and dorsum of hands. [2],[5],[7] It is more frequent in light-skinned individuals and affects predominantly males in the ratio of 3:1. [7],[8]

The present report document a case of KA of lip that misled us to a clinical diagnosis of squamous cell carcinoma because of its clinical appearance and concomitant presence of bilateral leukoplakia of the buccal mucosa. Crucial role of histopathology is highlighted in such clinical entities because of drastic difference in their overall management and prognosis.


   Case Report Top


A 50-year male patient reported to the Out Patient Department of Oral Medicine and Radiology of I.T.S. Centre for Dental Studies and Research, Muradnagar, India, with the chief complaint of a painless lip ulceration for the last one and half month. He stated that the lesion that appeared six weeks earlier started as a small hard papule with a central depression, and it grew rapidly for the first 4 weeks and then appeared to have stabilized. There was no description of any previous local trauma. However, patient reported continuous out-door activities in sun to earn his livelihood and history of heavy smoking for the past 20 years. His medical history was otherwise unremarkable.

The patient was moderately built and nourished for his age with all vital signs within normal limits. The local examination revealed a red and gray, raised, well circumscribed, sessile, nodular lesion measuring 2 cm in its greatest dimension with raised edges at the vermilion border of the lower lip on the left side of the face [Figure 1]. The center of the growth was slightly umblicated, erythematous and filled with a yellow cheesy material and the periphery appeared stippled with brown crustations. There was absence of associated regional lymphadenopathy as well as tenderness, bleeding and purulent discharge. Intraoral examination revealed presence of grayish white keratotic patch on both sides of buccal mucosa measuring 1.5 cm × 1.5 cm with an extension from the corner of the mouth to the second premolar region. Surface characteristically revealed corrugated appearance even on stretching the mucosa with absence of bleeding points or associated tenderness on palpation. Based on the clinical appearance and significant tobacco habit a provisional diagnosis of squamous cell carcinoma in an existing leukoplakia was made.
Figure 1: Clinical picture

Click here to view


For confirmation, an excisional biopsy for lip lesion and an incisional biopsy from left buccal mucosa were performed. The histopathological examination of the lip examination showed hyperplastic stratified squamous epithelium covered with thick layer of parakeratin. Surface showed characteristic parakeratin plugging. Invasive front was regular and typically invading in pseudo-carcinomatous formation [Figure 2]. Underlying connective tissue was normal with sebaceous glands and salivary glands, which were not infiltrated. The pushing front of the lesion showed epithelial islands with the connective tissue responding by an inflammatory infiltrate. Dysplasia was absent. Margins of the lesion showed a classical change of normal epithelial stratified epithelium to the abrupt change of the lesional tissue without any evidence of malignancy suggesting features of KA [Figure 3].
Figure 2: Section showing epithelial islands invading in pseudo carcinomatous fashion. Parakeratin plugging is clearly evident. [H and E, 4× magnification]

Click here to view
Figure 3: Section showing abrupt change from normal stratified epithelium to the lesional tissue of keratoacanthoma. [H and E, 4× magnification]

Click here to view


Lesion from the buccal mucosa on histopathological examination showed hyperplasic stratified squamous epithelium exhibiting hyperorthokeratosis. The underlying connective tissue showed melanosomes and melanophages in abundance intermingling with the chronic inflammatory cell infiltrate suggestive of Leukoplakia without dysplasia and associated with focal melanosis. Secondary healing occurred uneventfully at both biopsy sites over a period of 4 weeks. After a 3-month follow-up, there was no evidence of reoccurrence of the lesion.


   Discussion Top


KA is a relatively common skin lesion. Although cutaneous KAs are considered to arise from hair follicles, the rare cases reported occurring on mucous membranes would suggest possible origin from surface epithelium. [3],[7] Sir Jonathan Hutchinson in 1889 provided the first description of the KA characterizing it as the "crateriform ulcer of the face," a form of acute epithelial cancer. Freudenthal was, however, credited with coining the name "KA," which was employed by Rook and Whimster (1950) in their report of 29 cases. Synonyms for the KA include Kyste Sebace Atypique, Molluscum Pseudo Carcinomatosum, Verrucome Avec Adenite, self-healing primary squamous carcinoma, tumor like keratosis, multiple self-healing epithelioma of the skin, and keratocarcinoma. [9] Although most early descriptions emphasized the solitary keratacanthoma; [7] the multiple familial type of KAs were first described by Ferguson Smith in 1934 and the generalized eruptive type was described by Marian Grzybowski in 1950. [9],[10]

Ghadially et al.[11] conducted a study on the etiology of KA and concluded that acitinic factor, [7],[8],[9],[11],[12],[13],[14],[15],[16] Chemical carcinogens like pitch tar, coal and mechanic oil, [7],[9],[11] trauma [6] genetics, [17],[18] and virus [6],[17],[11],[19] were a contributory factor for this lesion. KA of oral cavity and lips in the Middle East is known to occur in smokers of "shisha" and "Goza," which contains a mixture of crude tobacco fermented with molasses and fruits." [20] Other postulated etiologic factors include chemotherapy, [21] circulating carcinogenic factors and immunologic factors. [7],[19]

KAs are commonly found on the skin exposed to sunlight including the vermilion border of the lower lip but have also been reported on the mucous membrane of the conjuctiva, glans penis, and oral cavity. [1],[3],[5],[8],[11],[17],[22],[23],[24],[25] History of occupational sun exposure and long history of heavy smoking might be contributory in the present case.

KA has been reported in patients ranging from 14 to 86 years, the average age being 56 years for both males and females. Eighty percent of the patients were over 40 years of age, and the maximum incidence was in the 50-70 year age group, with the male to female ratio of 2:1. [11] The multiple KA of Ferguson Smith has its onset in adolescence. [9],[12] Veidenheimer and Fidler estimated that keratocanthomas were one-third as common as squamous cell carcinomas, while Linell et al. in 1956 claimed that 20% of tumors formerly diagnosed as squamous cell carcinoma turned out to be KAs. [12]

There are three clinical stages in the natural history of KA as follows.

  1. Proliferative: A rapidly enlarging papulo nodule.
  2. Mature: A bud or hemisphere-shaped tumor with central keratinous core.
  3. Involutional or resolving: Akeratotic necrotic tumor, leaving a puckered hypo-pigmented scar. [7],[9],[22],[26] The histogenesis of KA was first reported by MacCormic and Schaff, who suggested that the development of this lesion was attributable to hypertrophic and inflammatory, changed in sebaceous cysts. However, now the probable derivation of the cutaneous KAs from the hair follicles and of the intraoral KAs from the sebaceous glands is well accepted. [7],[24] Ghadiallys studies classified KA into three morphologic types: type 1, bud-shaped; type 2, or dome-shaped; type 3 or berry-shaped. [7],[9],[22] The latter two are considered to be of lower follicular origin and the former is evaluated as of upper follicular origin. The lesion presented in the case report was the berry type.


Apart from the solitary and multiple forms of KA, it also has several special morphologic and syndromic forms, viz., (i) agglomerate KA, (ii) KA centrifugum also known as coral-reef KA, (iii) giant KA, (iv) subungal KA, (v) intraoral and other mucous membrane KAs which can occur on the hard palate, lips and other oral sites. Because there are no hair follicles in oral mucosa, the KA may develop from ectopic sebaceous glands, (vi) multiple KAs of Ferguson Smith types (vii) multiple persistent KAs, viii) generalized eruptive KAs of Grzybowski, (ix) KAs in Torre syndrome, (x) KA in xeroderma pigmentosum, (xi) KAs in nevus sebaceous of Jodassohn. [7],[9],[10],[27]

The most important and frequent consideration in the clinical and histopathological differential diagnosis is SCC. Fortunately the morphological features and growth pattern of a KA are sufficiently distinctive to be diagnosed in most cases, KA tends to be both exophytic and endophytic with a central keratin-filled crater, whereas cutaneous SCC is mainly endophytic with ulceration often present. The crater is surrounded by overhanging epithelial "lips" which are absent in the SCC. Intraepidermal abscess are common in KA and rarely seen in SCC. In the present case, a non-healing ulcer of more than 6 weeks duration with typical bony-shape surface characterizes along with concomitant presence of bilateral leukoplakia led to the formulation of squamous cell carcinoma in clinical diagnosis. In such a clinical situation, histopathology is mainstay in diagnosis, which will confirmatively differentiates KA from squamous cell carcinoma.

The case discussed here showed the characteristic appearance histopathologically in that the normal adjacent epithelium was elevated at the margins of the lesion, with an abrupt change from normal stratified epithelium to the lesion showing a hyperplastic-stratified squamous epithelium exhibiting parakeratin plugging. The epithelial islands were invading in pseudocarcinomatous fashion and walled-off by an inflammatory infiltrate. In addition, the base of the lesion was at the level of the sebaceous glands and not beyond it. A very important factor that has to be considered for the histopathological diagnosis to be accurate is that adequate amount of marginal tissue should always be included in the biopsy. Since an excisional biopsy was performed with sufficient adjacent normal tissue, the histopathological diagnosis was confirmed in this case.

Other clinical differential diagnosis besides SCC includes verrucous carcinoma, giant condyloma acuminatum of Buschke and Lowenstein, hypertrophic acitinic keratosis, warty dyskeratoma, giant molluscum contagiosum, verruca vulgaris, metastatic cancer and deep fungal infection such as chromoblastomycosis. Small KAs seen in multiple eruptions may resemble Dariers disease.

Although the natural course of a KA is spontaneous regression, therapeutic regression is mandatory to differentiate it from SCC. Solitary KAs respond well to surgical excision. Excisional biopsy was performed in the present case as the size of the lesion was smaller in overall dimensions. Patient is still on follow-up without any evidence of recurrence. Other therapies used are cryotherapy with liquid nitrogen, electro dissection and curettage, radiation therapy, CO 2 laser surgery have been used in small KAs. Intralesional or topical treatment with 5-fluorouracil, corticosteroid, bleomycin, interferon alpha-2b, and methotrexate have also been used. [2],[7],[9],[15],[23],[26],[28]


   Summary Top


KA is a relatively common cutaneous lesion commonly arising from hair follicles. Moreover, its occurrence on mucous membranes would suggest possible origin from surface epithelium. This paper reports a case of KA localized on the prolabium of the lower lip. Attention is focused on the difficulty of making a correct diagnosis and on the complexity of differential diagnosis with squamous cell carcinoma as the patient also presented with bilateral leukoplakia on the buccal mucosa and with a long-standing smoking history. A complete excision was carried out, and the histopathological examination of the entire neoplastic mass ruled out the presence of squamous cell carcinoma.

 
   References Top

1.Freedman PD, Kerpel SM, Begel H, Lumerman H, Jamaica NY. Solitary Intraoral Keratoacanthoma. Oral Surg 1979;4:74-6.  Back to cited text no. 1
    
2.Kingman J, Callen JP. Keratoacanthoma. Arch Dermatol 1984;120:736-40.  Back to cited text no. 2
[PUBMED]  [FULLTEXT]  
3.Whyte AM, Hansen LS. The Intraoral Keratoacanthoma: A diagnostic problem. Brit J Oral and Maxillo Surg 1986;24:438-41.  Back to cited text no. 3
    
4.Eversole LR, Leider SA, Alexander G. Intraoral and Labial Keratoacanthoma. Oral Surg 1982;54:663-7.  Back to cited text no. 4
    
5.Svirsky JA, Freedman PD, Lumerman H. Solitary Intraoral keratoacanthoma. Oral Surg 1977;43:116-22.  Back to cited text no. 5
[PUBMED]    
6.Pattee SF, Silvis NG. Keratoacanthoma developing in sites of previous trauma. J Am Acad Dermatol 2003;48:s35-8.  Back to cited text no. 6
[PUBMED]  [FULLTEXT]  
7.Schwartz RA. The Keratoacanthoma: A Review. J Surg Oncol 1979;12:305-17.  Back to cited text no. 7
[PUBMED]    
8.Havel G, Regan BO, Essing A, Donath K. Intraoral Keratoacanthoma: An eruptive variant and review of literature. Brit Dent J 1991;170:336-9.  Back to cited text no. 8
    
9.Schwartz RA. Keratoacanthoma. J Am Acad Dermatol 1994;30:1-19.  Back to cited text no. 9
[PUBMED]    
10.Yell JA. Grzybowski generalized eruptive keratoacanthoma. J Ryl Sci Med 1991;84:170-1.  Back to cited text no. 10
    
11.Ghadially FN, Barton BW, Kerridge DF. The etiology of Keratoacanthoma. Cancer 1963;16:603-11.  Back to cited text no. 11
[PUBMED]    
12.Katoulis AC, Bozi Evangeli. Multiple Keratoacanthoma Ferguson Smith type. Orphanet Encycl 2005. http://www.orpha.net/data/patho/GB/uk-Keratoacanthoma.pdf   Back to cited text no. 12
    
13.Hoqal IA, Bhatt TA. Keratoacanthoma: An Unusual Presentation. Bahrain Med Bull 2009;1:31.  Back to cited text no. 13
    
14.Ahn SK, Lee HS, Han SK, Lee SH, Lee S. Multiple Basal Cell Carcinoma associated with keratoacanthoma. Yonsei Med J 1992;3:277-80.  Back to cited text no. 14
    
15.Ramos LM, Cardoso SV, Loyola AM, Rocha MA, Durighetto-Júnior AF. Keratoacanthoma of the inferior lip: Review and report with spontaneous regression. J Appl Oral Sci 2009;17:262-5.  Back to cited text no. 15
    
16.Kraemer KH. Sunlight and skin cancer. Proc Natl Acad Sci USA 1997;94:11-4.  Back to cited text no. 16
[PUBMED]  [FULLTEXT]  
17.Heideureich RK, Gongloff RK, Weskott WB. A Solitary Exophytic lesion on Mandibular retromolar gingival. J Am Dent Ass 1986;112:377-9.  Back to cited text no. 17
    
18.Sarabi K, Selim A, Khachemoune A. Sporadic and Syndromic Keratoacanthomas: Diagnosis and Management. Dermatol Nurs 2007;19:166-70.  Back to cited text no. 18
[PUBMED]    
19.Saftic M, Batinac T, Zamolo G, Coklo M, Simat M, Mustac E, et al. HPV 6 - Positive Giant Keratoacanthoma in an Immunocomprimised Patient. Tumori 2006;92:79-82.  Back to cited text no. 19
[PUBMED]    
20.Ibrahim E, Mirghan EL. Squamous Cell Carcinoma and Keratoacanthoma of the lower lip associated with Goza and Shisha smoking. Int J Dermatol 1999;38:108-10.  Back to cited text no. 20
    
21.Marquez BC, Smithberger EE, Beir SM. Multiple Keratoacanthoma arising in the setting of Sorafenib therapy: Novel Chemoprophylaxis with Bexarotone. Cancer Control 2009;16:66-9.  Back to cited text no. 21
    
22.Leibovitch I, Huilgol SC, James CL, Hsuan JD, Davis G, Selva D. Periocular Keratoacanthoma: Can we rely on the clinical diagnosis ?. Br J Opthalmol 2005;89:1201-4.  Back to cited text no. 22
    
23.Chen YK, Lin ML, Lein CC, Chen CH. Keratoacanthoma of the tongue: A diagnostic problem. Otolaryngol Head Neck Surg 2003;128:581-2.  Back to cited text no. 23
    
24.Janelte A, Pecaro Bernard P, Lonergan M, Lingen MW. Solitary Intraoral Keratoacanthoma. J Oral Maxillo Surg 1996;54:1026-30.  Back to cited text no. 24
    
25.Berrone S, Gionnie PP, Gallesio C. Keratoacanthoma of lower lip. Minerva Stomatol 1992;41:597-601.  Back to cited text no. 25
    
26.Karaa A, Kachemoune A, Usatine RP. Tumour with central crusting. J Fam Pract 2008;57:669-71.  Back to cited text no. 26
    
27.Nelson LM, Barbara S. Self healing psudo cancers of the skin. California Med 1959;90:49-54.  Back to cited text no. 27
    
28.Jackson R. Keratoacanthoma. Can Med Assoc J 1959;80:551-2.  Back to cited text no. 28
[PUBMED]  [FULLTEXT]  


    Figures

  [Figure 1], [Figure 2], [Figure 3]

This article has been cited by
1 Keratoacanthoma Clinical Behavior
Jacqueline A. Savage,John C. Maize
The American Journal of Dermatopathology. 2014; 36(5): 422
[Pubmed] | [DOI]



 

Top
Print this article  Email this article
 
 
  Search
 
  
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Article in PDF (1,427 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  


    Abstract
   Introduction
   Case Report
   Discussion
   Summary
    References
    Article Figures

 Article Access Statistics
    Viewed3815    
    Printed112    
    Emailed0    
    PDF Downloaded86    
    Comments [Add]    
    Cited by others 1    

Recommend this journal