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Oral Linear Epidermal Nevus: A Review of the Literature and Report of Two New Cases

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Abstract

Linear epidermal nevus (LEN) is a sporadic hamartomatous lesion of the skin due to the proliferation of clones of embryonic ectodermal cells, which are arranged according to a typical linear configuration known as Blaschko’s lines. Oral involvement of LEN is very rare and few cases have been reported in the medical literature. We report two new cases of LEN with exclusive oral involvement, which presented with the typical unilateral or midline distribution. Oral LEN presents as an exophytic lesion with well defined borders and a verrucous or papillary surface which correspond, histologically, to epithelial papillomatosis with a moderate degree of hyperkeratosis and acanthosis. Oral LEN appears mainly at birth, grows slowly during childhood and stabilize by adolescence. Localized lesions do not show any recurrence after surgical removal but widespread lesions seem to have more risk of recurrence and are more difficult to manage. Functional problems and malignant transformation have not been reported.

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References

  1. Thomas VD, Swanson NA, Lee KK. Benign epithelial tumors, hamartomas and hyperplasias. In: Wolff K, Goldsmith LA, Katz SI, Gilchrest BA, Pallers AS, Lefell DJ, et al., editors. Fitzpatrick’s. dermatology in general medicine. 7th ed. New York: Mc Graw Hill; 2008. p. 1057.

    Google Scholar 

  2. Happle R, Assim A. The lines of Blaschko on the head and neck. J Am Acad Dermat. 2001;44:612–4.

    Article  CAS  Google Scholar 

  3. Bolognia JL, Orlow JJ, Glick SA, et al. Lines of Blaschko. J Am Acad Dermat. 1994;31:157–90.

    Article  CAS  Google Scholar 

  4. Happle R. Epidermal nevus syndromes. Semin Dermatol. 1995;14:111–21.

    Article  CAS  PubMed  Google Scholar 

  5. Brown HM, Gorlin RJ. Oral mucosal involvement in nevus unius lateris (ichthyosis histrix). Arch Dermatol. 1960;81:61–7.

    Google Scholar 

  6. Hickman RE, Eveson JW, Cawson RA. Nevus unius lateris and intraoral verrucous nevi. Oral Surg Oral Med Oral Pathol. 1988;66:226–9.

    Article  CAS  PubMed  Google Scholar 

  7. Coley-Smith A, Shaw L. Oral findings in patients with epidermal nevi: a report of two cases. Dent Update. 1996;23:158–60.

    CAS  PubMed  Google Scholar 

  8. Ozçelik D, Haydar-Parlak A, Ozturk A, et al. Unilateral linear verrucous epidermal nevus of the face and oral mucosa. Plast Reconstr Surg. 2005;115:17e–9e.

    Article  PubMed  Google Scholar 

  9. Haberland-Carrodeguas C, Allen CM, Lovas JGL, et al. Review of linear epidermal nevus with oral mucosal involvement—series of five new cases. Oral Dis. 2008;14:131–7.

    Article  CAS  PubMed  Google Scholar 

  10. Naylor MF. Benign epithelial tumors and hamartomas. In: Sams WM, Lynch PJ, editors. Principles and practise of dermatology. 2nd ed. New York, NY: Churchill Livingstone; 1996. p. 215–6.

    Google Scholar 

  11. Alsaleh QA, Nanda A, Hassab-El-Naby HM, et al. Familial inflammatory linear verrucous epidermal nevus (ILVEN). Int J Dermatol. 1994; 33–52.

  12. Happle R. Transposable elements and lines of Blaschko: a new perspective. Dermatology. 2002;204:4–7.

    Article  CAS  PubMed  Google Scholar 

  13. Warnke PH, Schimmelpenning GW, Happle R, et al. Intraoral lesions associated with sebaceous nevus syndrome. J Cutan Pathol. 2006;33:175–80.

    Article  CAS  PubMed  Google Scholar 

  14. Baghaei-Rad M, Doku C, Ficarelli J. Epidermal nevus syndrome with maxillary involvement. J Oral Maxillofac Surg. 1982;40:821–3.

    Article  CAS  PubMed  Google Scholar 

  15. Basopoulou-Kyrkanidou E, Alexanridis C, Tosious KI, et al. Epidermal nevus syndrome with development of a mandibular ameloblastoma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2000;90:64–70.

    Article  Google Scholar 

  16. Kaplan I, Metzker A, Calderon S. Epidermal nevus syndrome with maxillary involvement. Int J Oral Maxillofac Surg. 1993;22:298–300.

    Article  CAS  PubMed  Google Scholar 

  17. Kelley JE, Hibbard ED, Giansanti JS. Epidermal nevus syndrome. Report of a case with unusual oral manifestations. Oral Surg Oral Med Oral Pathol. 1972;34:774–80.

    Article  CAS  PubMed  Google Scholar 

  18. Ichikawa T, Saiki M, Kaneko M, et al. Squamous cell carcinoma arising in a verrucous epidermal nevus. Dermatology. 1996;193:135–8.

    Article  CAS  PubMed  Google Scholar 

  19. Michel JL, Has C, Has V. Resurfacing CO2 laser treatment of linear epidermal nevus. Eur J Dermatol. 2001;11:436–9.

    CAS  PubMed  Google Scholar 

  20. Fox BJ, Lapins NA. Comparison of treatment modalities for epidermal nevus: a case report and review. J Dermatol Surg Oncol. 1983;9:879–85.

    CAS  PubMed  Google Scholar 

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Correspondence to Giuseppe Ficarra.

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Tesi, D., Ficarra, G. Oral Linear Epidermal Nevus: A Review of the Literature and Report of Two New Cases. Head and Neck Pathol 4, 139–143 (2010). https://doi.org/10.1007/s12105-010-0165-7

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  • DOI: https://doi.org/10.1007/s12105-010-0165-7

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