Buruli ulcer is an infectious necrotizing panniculitis due to Mycobacterium ulcerans which heals leaving scars. On these scars, squamous cell carcinoma may occur in the long term, even in case of skin grafting. We report a case of squamous cell carcinoma occurring on a directed buruli ulcer scar.

Observation: A 35-year-old patient with a history of buruli ulcer healed with a skin graft in a specialized center for about 13 years, consulted for a cauliflower-like ulcerating swelling on the left elbow. The examination showed a large ulcerating cauliflower-like swelling. The diagnosis of squamous cell carcinoma was retained, and an amputation was performed without chemotherapy. There was no recurrence after six months of follow-up.

Conclusion: After a good healing, Buruli ulcer seems to present a risk of long-term evolution towards cancer. This observation raises the question of the carcinogenic role of mycobacterium ulcerans.

Buruli ulcer; Scar; Squamous cell carcinoma.

Almamy D, Ida Aurélie LL, Armel OM, Akanbi SM, Irené G, et al. (2022) Squamous Cell Carcinoma On Buruli Ulcer Graft Scar, Ivory Coast. J Dermatolog Clin Res 10(1): 1148.

Buruli ulcer is an infectious necrotizing panniculitis caused by Mycobacterium ulcerans [1]. Currently, the endemic continues to grow and its incidence is increasing dramatically, especially in West African countries such as Ivory Coast. Buruli ulcer is characterized by its chronic evolution, characterized by extensive skin eruptions complicated by dystrophic, fibrous and retractile scars [2,3,5]. On the other hand, directed healing gives considerable scars that are resistant to traumatic events. On these fibrous scars, squamous cell carcinoma may occur in the long term. In Abidjan, the first case was observed in 2010 [6], then eight cases were observed in 2015 [1] and finally one case in Bouaké in 2019. However, there is no on directed scars. We report a case of squamous cell carcinoma occurring on directed scarring of buruli ulcer in a 35-year-old patient without comorbidity.

A 35-year-old HIV-negative patient with a history of buruli ulcer healed with a skin graft in a specialized buruli ulcer management center in Kongouanou (Yamoussoukro) for about 13 years, consulted for an ulcerating swelling on the left elbow for the past 2 months. Examination showed a large swelling of about 12 cm in diameter, ulcerating and bubbling, with a cauliflower-like appearance, bleeding easily on contact, painful, and located on the inner side of the left elbow (Figure 1a,1b).

Figure 1 A and B: Burgeoning ulcer tumor

The peri-lesional skin was normal in appearance. Biology revealed a normocytic hypochromic anemia. Histology revealed a proliferation of atypical squamous cells (large hyperchromatic nuclei, numerous mitoses) in invasive lobules, associated with disorders of keratinization. Finally, the tumor stroma is inflammatory (Figure 2).

Figure 2 Histological appearance of squamous cell carcinoma.

X-ray of the elbow showed bone lysis. The diagnosis of squamous cell carcinoma was retained without metastasis. Amputation was performed without chemotherapy. And there was no recurrence after six months of follow-up.

Buruli ulcer is hyperendemic in West and Central Africa : 16,517 cases were recorded from 2006 to 2015 and each year, about 500 new cases are recorded in Côte d’Ivoire, which remains a very active focus [4]. The epidemiology of the infection responsible for scarring [5,7] explains the young age of our patient and the location of the carcinoma on the limb. No comorbidity, in particular HIV infection, which is a factor favoring the development of malignant tumors in sub-Saharan Africa, was noted in our observation. Healing of the lesions occurred after several months of treatment. In our report, our patient had very good quality scars after directed healing. The occurrence of cancer in buruli ulcer scars is known [1,6,14].

Isolated cases of squamous cell carcinoma have already been described [8,9]. The first Ivorian observation was reported in 2010 [6]. Since then, eight cases have been recruited by the Abidjan center, which suggests a higher number at the national level since the Abidjan center does not have a monopoly on Buruli ulcer management. All these cases developed on fibrous and retractile scars. If sun exposure is the main risk factor for cutaneous squamous cell carcinoma in fair-skinned people, nonsun factors would be involved in people with pigmented skin. These are mainly chronic leg ulcers (neglected post-traumatic or  Buruli ulcer in order to detect the first signs of carcinomatous degeneration and to sensitize the patients to an early consultation in front of any modification of their scars. This carcinological prevention requires very early management (at a stage without bone involvement or metastasis) of cases, could improve their prognosis.

After a complete healing, Buruli ulcer appears to have a longterm risk of progression to cancer. The scars of this condition, which could be considered as precancerous lesions. This observation calls into question the safety of directed healing or the carcinogenic role of mycobacterium ulcerans.

1. M Kaloga, A Diabaté, HS Kourouma, IP Gbery, A Sangaré, EJ Elidjé, et al. [Squamous cell carcinoma secondary to Buruli ulcer in West Africa]. Ann Derm Vene. 2016; 143: 16-20.

2. Pradinaud L, Couppié P, Versapuech J. Environmental skin mycobacteria including Mycobacterium ulcerans (“Buruli ulcer”) infection. EMC Maladies infectieuses. 2003.

3. B Saka, DE Landoh, B Kobara, KE Djadou, I Yaya, KB Yékplé, et al. [Profile of Buruli ulcer treated at the National Reference Centre of Togo: a study of 119 cases]. Bull Soc Pathol Exot. 2013; 106: 32-6.

4. Ecra E, Yoboue P, Aka B, Gbery I, Sangare A, Kanga K, et al. Complications of Buruli ulcer: analysis of 97 cases. Med Afr Noire. 2001; 48: 4.

5. Kanga JM, Kacou ED, Kouamé K, Kassi K, Kaloga M, Yao JK, et al. [Fighting against Buruli ulcer: the Côte-d’Ivoire experience]. Bull Soc Pathol Exot. 2006; 99: 34-8.

6. Kassi K, Kouame K, Allen W, Kouassi LA, Ance W, Kanga JM. Squamous cell carcinoma secondary to Buruli ulcer: a clinical case report in a young girl. Bacteriol Virusol Parazitol Epidemiol. 2010; 55: 25-8.

7. Nienhuis WA, Stienstra Y, Thompson WA, Awuah PC, Abass KM, Tuah W, et al. Antimicrobial treatment for early, limited Mycobacterium ulcerans infection: a randomised controlled trial. Lancet. 2010; 375:664-72.

8. Minutilli E, Orefici G, Pardini M, Giannoni F, Muscardin LM, Massi G, et al. Squamous cell carcinoma secondary to Buruli ulcer. Dermatol Surg. 2007; 33:872-875.

9. Evans MR, Etuaful SN, Amofah G, Adjei O, Lucas S, Wansbrough Jones MH. Squamous cell carcinoma secondary to Buruli ulcer. Trans R Soc Trop Med Hyg. 1999; 93:63-4.

10. Saka B, Souley Z, Kombaté K, Mouhari·Toure A, Akakpo S, Napo Koura G, et al. Les cancers cutanés au Togo: 223 observations. Med Trop. 2010; 70: 16971.

11. Dieng MT, Diop NN, Déme A, Sy TN, Niang 50, Ndiaye B. [Squamous cell carcinoma on black skin: 80 cases]. Ann Dermatol Venereol. 2004; 131: 1055-1057.

12. Ouahbi S, Droussi H, Boukind S, Dlimi M, Elatiqi OK, Elamrani MD, et al. Ulcère de Marjolin: complication redoutable des séquelles de brûlures. Ann Burns Fire Disasters. 2013; 26:199-204.

13. Wallingford SC, Olsen CM, Plasmeijer E, Green AC. Skin cancer arising in scars: a systematic review. Dermatol Surg. 2011; 37:1239-44.

14.Almamy Diabaté, Koffi Kouamé Pacôme Gbandama, Amon Anderson Stephen Kouabenan, Irenée Gué, Bamba Vagamon. A case of squamous cell carcinoma occurring on a scar of Buruli ulcer in Bouake, Ivory Coast. Pan Afr Med J. 2019; 33: 246.