Skip to main content

Advertisement

Log in

Marjolin ulcers after cultured epidermal autograft in severely burned patients: a rare case series and literature review

  • Clinical report
  • Published:
European Journal of Dermatology

Abstract

Background

The advent of tissue engineering and the clinical applications with cultured epidermal autograft (CEA) have improved the prognosis of severely burned patients. Marjolin ulcers (MUs) are a well-known complication of burns. These malignant neoplasm transformations of burn scars are usually squamous cell carcinomas with a higher incidence of regional metastases. Radical surgery remains the treatment of choice.

Objectives

To identify cases of malignant transformation occurring at sites of CEA in a cohort of 68 massively burned patients.

Materials & Methods

A retrospective single-centre study was performed from April 2017 to June 2019 at the Military Hospital of Clamart (France). A total of 34 patients treated between 1991 and 2013 (including one post-mortem) were included.

Results

Four cases of squamous cell carcinoma occurred in areas previously covered by CEA. The data from clinical and histopathological examination as well as treatment modalities are presented. One patient died as a result of the evolution of his squamous cell carcinoma, and two others required salvage amputation due to locoregional recurrence. The prevalence of these CEA-MUs is estimated at between 5.9% and 11.7% and the person-time incidence rate of CEA-related MUs is about 5.9 per 1,000 persons-years. In our study, the average time to malignant transformation seems considerably shortened (32–35 years for “classic burn MU” versus 15.7 years for CEA-MU).

Conclusion

This first documented case series of CEA-MUs demonstrates the need to inform caregivers and educate patients. Further studies are needed to specify the true incidence of CEA-graft site malignancy.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Similar content being viewed by others

References

  1. Copcu E. Marjolin’s ulcer: a preventable complication of burns? Plast Reconstr Surg 2009; 124: 156e–64e.

    Article  CAS  PubMed  Google Scholar 

  2. Dupree MT, Boyer JD, Cobb MW. Marjolin’s ulcer arising in a burn scar. Cutis 1998; 62: 49–51.

    CAS  PubMed  Google Scholar 

  3. Fleming MD, Hunt JL, Purdue GF, Sandstad J. Marjolin’s ulcer: a review and reevaluation of a difficult problem. J Burn Care Rehabil 1990; 11: 460–9.

    Article  CAS  PubMed  Google Scholar 

  4. Copcu E, Aktas A, Sişman N, Oztan Y. Thirty-one cases of Marjolin’s ulcer. Clin Exp Dermatol 2003; 28: 138–41.

    Article  CAS  PubMed  Google Scholar 

  5. Yu N, Long X, Lujan-Hernandez JR, et al. Marjolin’s ulcer: a preventable malignancy arising from scars. World J Surg Oncol 2013; 11: 313.

    Article  PubMed  PubMed Central  Google Scholar 

  6. Pratt DV, Pelton RW, Patel BC, Anderson RL. Burn scar malignancies of the eyelids. Ophthal Plast Reconstr Surg 2000; 16: 432–7.

    Article  CAS  PubMed  Google Scholar 

  7. Kadir AR. Burn scar neoplasm. Ann Burns Fire Disasters 2007; 20: 185–8.

    CAS  PubMed  PubMed Central  Google Scholar 

  8. Bozkurt M, Kapi E, Kuvat SV, Ozekinci S. Current concepts in the management of Marjolin’s ulcers: outcomes from a standardized treatment protocol in 16 cases. J Burn Care Res 2010; 31: 776–80.

    Article  PubMed  Google Scholar 

  9. Bean DJ, Rees RS, O’Leary JP, Lynch JB. Carcinoma of the hand: a 20-year experience. South Med J 1984; 77: 998–1000.

    Article  CAS  PubMed  Google Scholar 

  10. Ozek C, Celik N, Bilkay U, Akalin T, Erdem O, Cagdas A. Marjolin’s ulcer of the scalp: report of 5 cases and review of the literature. J Burn Care Rehabil 2001; 22: 65–9.

    Article  CAS  PubMed  Google Scholar 

  11. Kerr-Valentic MA, Samimi K, Rohlen BH, Agarwal JP, Rockwell WB. Marjolin’s ulcer: modern analysis of an ancient problem. Plast Reconstr Surg 2009; 123: 184–91.

    Article  CAS  PubMed  Google Scholar 

  12. Cantwell P, Brooks A. Multiple melanoma in a burns scar. BMJ Case Rep 2018; 11: e227295.

    Article  PubMed  PubMed Central  Google Scholar 

  13. Gül U, Kiliç A. Squamous cell carcinoma developing on burn scar. Ann Plast Surg 2006; 56: 406–8.

    Article  PubMed  Google Scholar 

  14. Abdi MA, Yan M, Hanna TP. Systematic review of modern case series of squamous cell cancer arising in a chronic ulcer (Marjolin’s ulcer) of the Skin. JCO Glob Oncol 2020; 6: 809–18.

    Article  PubMed  Google Scholar 

  15. Khan K, Schafer C, Wood J. Marjolin ulcer: a comprehensive review. Adv Skin Wound Care 2020; 33: 629–34.

    Article  PubMed  Google Scholar 

  16. Theopold C, Hoeller D, Velander P, Demling R, Eriksson E. Graft site malignancy following treatment of full-thickness burn with cultured epidermal autograft. Plast Reconstr Surg 2004; 114: 1215–9.

    Article  PubMed  Google Scholar 

  17. Theopold C, Eriksson E. The need for aggressive follow-up after cultured epidermal autograft-grafted full-thickness burn. Plast Reconstr Surg 2006; 117: 708.

    Article  CAS  PubMed  Google Scholar 

  18. Hickerson WL, Remmers AE, Recker DP. Twenty-five years’ experience and beyond with cultured epidermal autografts for coverage of large burn wounds in adult and pediatric patients, 1989–2015. J Burn Care Res 2019;40:157–65.

    Article  PubMed  Google Scholar 

  19. Rheinwald JG, Green H. Serial cultivation of strains of human epidermal keratinocytes: the formation of keratinizing colonies from single cells. Cell 1975; 6: 331–43.

    Article  CAS  PubMed  Google Scholar 

  20. Cuono C, Langdon R, McGuire J. Use of cultured epidermal autografts and dermal allografts as skin replacement after burn injury. Lancet Lond Engl 1986; 1: 1123–4.

    Article  CAS  Google Scholar 

  21. Cuono CB, Langdon R, Birchall N, Barttelbort S, McGuire J. Composite autologous-allogeneic skin replacement: development and clinical application. Plast Reconstr Surg 1987; 80: 626–37.

    Article  CAS  PubMed  Google Scholar 

  22. O’Connor N, et al. Grafting of burns with cultured epithelium prepared from autologous epidermal cells. Lancet Lond Engl 1981; 1: 75–8.

    Article  Google Scholar 

  23. Atiyeh BS, Costagliola M. Cultured epithelial autograft (CEA) in burn treatment: three decades later. Burns J Int Soc Burn Inj 2007; 33: 405–13.

    Article  Google Scholar 

  24. Paddle-Ledinek JE, Cruickshank DG, Masterton JP. Skin replacement by cultured keratinocyte grafts: an Australian experience. Burns J Int Soc Burn Inj 1997; 23: 204–11.

    Article  CAS  Google Scholar 

  25. Cirodde A, Leclerc T, Jault P, Duhamel P, Lataillade J-J, Bargues L. Cultured epithelial autografts in massive burns: a single-center retrospective study with 63 patients. Burns J Int Soc Burn Inj 2011; 37: 964–72.

    Article  Google Scholar 

  26. Teepe RG, Kreis RW, Koebrugge EJ, et al. The use of cultured autologous epidermis in the treatment of extensive burn wounds. J Trauma 1990; 30: 269–75.

    Article  CAS  PubMed  Google Scholar 

  27. Iqbal FM, Sinha Y, Jaffe W. Marjolin’s ulcer: a rare entity with a call for early diagnosis. BMJ Case Rep 2015; 2015: bcr2014208176.

    Article  PubMed  PubMed Central  Google Scholar 

  28. Metwally IH, Roshdy A, Saleh SS, Ezzat M. Epidemiology and predictors of recurrence of Marjolin’s ulcer: experience from Mansoura Universityxs. Ann R Coll Surg Engl 2017; 99: 245–9.

    Article  CAS  PubMed  Google Scholar 

  29. Xiao-Wu W, Herndon DN, Spies M, Sanford AP, Wolf SE. Effects of delayed wound excision and grafting in severely burned children. Arch Surg Chic Ill 2002; 137: 1049–54.

    Article  Google Scholar 

  30. Türegün M, Nişanci M, Güler M. Burn scar carcinoma with longer lag period arising in previously grafted area. Burns J Int Soc Burn Inj 1997; 23: 496–7.

    Article  Google Scholar 

  31. Lawrence EA. Carcinoma arising in the scars of thermal burns, with special reference to the influence of the age at burn on the length of the induction period. Surg Gynecol Obstet 1952; 95: 579–88.

    CAS  PubMed  Google Scholar 

  32. Das KK, Chakaraborty A, Rahman A, Khandkar S. Incidences of malignancy in chronic burn scar ulcers: experience from Bangladesh. Burns J Int Soc Burn Inj 2015; 41: 1315–21.

    Article  Google Scholar 

  33. Edwards MJ, Hirsch RM, Broadwater JR, Netscher DT, Ames FC. Squamous cell carcinoma arising in previously burned or irradiated skin. Arch Surg Chic Ill 1989; 124: 115–7.

    Article  CAS  Google Scholar 

  34. Costache M, Desa LT, Mitrache LE, et al. Cutaneous verrucous carcinoma — report of three cases with review of literature. Rom J Morphol Embryol 2014; 55: 383–8.

    PubMed  Google Scholar 

  35. Braye F, Oddou L, Bertin-Maghit M, et al. Widely meshed autograft associated with cultured autologous epithelium for the treatment of major burns in children: report of 12 cases. Eur J Pediatr Surg 2000; 10: 35–40.

    Article  CAS  PubMed  Google Scholar 

  36. Nthumba PM. Marjolin’s ulcers in sub-Saharan Africa. World J Surg 2010; 34: 2272–7.

    Article  PubMed  Google Scholar 

  37. Ozek C, Cankayali R, Bilkay U, et al. Marjolin’s ulcers arising in burn scars. J Burn Care Rehabil 2001; 22: 384–9.

    Article  CAS  PubMed  Google Scholar 

  38. Reich-Schupke S, Doerler M, Wollina U, et al. Squamous cell carcinomas in chronic venous leg ulcers. Data of the German Marjolin Registry and review. J Dtsch Dermatol Ges 2015; 13: 1006–13.

    PubMed  Google Scholar 

  39. Delaitre L, Martins-Héricher J, Truchot E, et al. [Regression of cutaneous basal cell and squamous cell carcinoma under pembrolizumab]. Ann Dermatol Venereol 2020; 147: 279–84.

    Article  CAS  PubMed  Google Scholar 

  40. Lavaud J, Blom A, Longvert C, Fort M, Funck-Brentano E, Saiag P. Pembrolizumab and concurrent hypo-fractionated radiotherapy for advanced non-resectable cutaneous squamous cell carcinoma. Eur J Dermatol 2019; 29: 63640.

    Google Scholar 

  41. Baus A, Chatard M, Bich CS, et al. [Current place of cultured epithelial autografts in the management of massive burns and future prospects: Literature review]. Ann Chir Plast Esthet 2021; 66: 10–8.

    Article  CAS  PubMed  Google Scholar 

Download references

Acknowledgment and disclosures

Acknowledgments: We would like to thank Dr. N’Guyen Anhtuan (Pathology Department, Begin Military Hospital, Saint Mandé, France), Dr. Benali-Abdeellaoui Khadija (Nuclear Medicine Department, Bichat Hospital, Paris, France), Dr Pinatel Baptiste (Plastic and Reconstructive Surgery, Saint Luc and Saint Joseph Hospital, Lyon, France), Dr Trouillas Marina (PhD, IRBA/French Armed-forces Biomedical Research Institute, Clamart, France) and Dr Jacob Louis (Physical and Rehabilitation Medicine Department, Garches Hospital, France) for their proofreading and scientific contribution.

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Arnaud Baus.

Additional information

Conflicts of interest

None of the authors has a financial interest in any of the products, devices or drugs mentioned in this manuscript. This review did not receive any funding. The opinions or assertions expressed here are the private views of the authors and are not to be considered as official or as reflecting the views of the French Defence Health Service.

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Baus, A., Keilani, C., Brunet-Possenti, F. et al. Marjolin ulcers after cultured epidermal autograft in severely burned patients: a rare case series and literature review. Eur J Dermatol 31, 759–770 (2021). https://doi.org/10.1684/ejd.2021.4174

Download citation

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1684/ejd.2021.4174

Key words

Navigation