Squamous cell carcinoma associated with an active cutaneous leishmaniasis in immunocompetent patient: case presentation of an unlikely association and literature—review

Abstract

Background Association between leishmaniasis and malignancy can be classified into four categories: leishmaniasis mimicking malignancy, leishmaniasis co-existing with malignancy, malignancy developing in patients with leishmaniasis scar, and leishmaniasis developing in patients with malignancy. In immunocompetent patients, the main form of association is cutaneous squamous cell carcinoma (cSCC) developing within cutaneous leishmaniasis scar years after cutaneous leishmaniasis is cured. Association of active cutaneous leishmaniasis and cSCC is exceptional, we are aware of two more cases. Case presentation A 30-year-old man presented with 2 years history of an unhealed wound on the dorsum nasi. As there still exist few sites of leishmaniasis in Morocco, systematic screening for leishmania was performed. Leishmania bodies were identified on slit skin smear by Giemsa staining. The patient received local antibiotic and on-site injections of 4 cc of meglumine antimonate for 2 months without any improvement. The lesion volume has increased significantly, a biopsy revealed an invasive squamous cell carcinoma. After staging assessment, the patient underwent a complete removal of the nasal tumor with a 1-cm margin, associated with right modified radical neck dissection. Histopathological examination confirmed the diagnosis of SCC with no lymph nodes metastasis. Nasal reconstruction was performed 2 weeks later using a frontal flap. Oncology meeting board advised adjuvant radiation on the tumor. The patient is followed up regularly and remains free of disease for a year now. Conclusion Although many cancers are related to infection (viral or parasitic), there is no proven link between leishmaniasis and malignancy. However, there are many etiopathogenic theories based on pathology finding that involve chronic inflammation inducing dysplasia, mitotic abnormalities, and expression of p53.

Current Challenges in the Association between Canine Leishmaniasis and Malignant Disorders

“…An unlikely association of squamous cell carcinoma with an active cutaneous leishmaniasis was reported in an immunocompetent human patient [25], and a report of a case of basal cell carcinoma (Clark level IV) that had been developed on characteristic active lesion of mucocutaneous leishmaniasis, suggesting that carcinogenesis, in this case, was triggered by Leishmania parasites and induced by sun's ultraviolet (UV) radiation exposure [56]. Although causality is still to be proved, a biopsy may be essential to rule out associated skin malignancy in reluctant cutaneous leishmaniasis [25]. A report of a solitary cutaneous leiomyoma mimicking leishmaniasis demonstrates that neoplasms mimicking cutaneous leishmaniasis are an alert for an appropriate differential diagnosis [57].…”

“…However, most of the evidence about the association between cutaneous leishmaniasis and skin cancer is still based on case reports and other clinical observations rather than strong epidemiological observational studies [55]. An unlikely association of squamous cell carcinoma with an active cutaneous leishmaniasis was reported in an immunocompetent human patient [25], and a report of a case of basal cell carcinoma (Clark level IV) that had been developed on characteristic active lesion of mucocutaneous leishmaniasis, suggesting that carcinogenesis, in this case, was triggered by Leishmania parasites and induced by sun's ultraviolet (UV) radiation exposure [56]. Although causality is still to be proved, a biopsy may be essential to rule out associated skin malignancy in reluctant cutaneous leishmaniasis [25].…”

“…Among the proposed theories, the chronic inflammation induced by Leishmania persistence, the interference with the local systemic immune system, and a direct implication of the parasite in the pathogenesis of cancer are considered most important. Etiopathogenic theories are based on the pathological observation that chronic inflammation can lead to development of dysplasia, abnormalities in cell division and activation of the p53 protein (an important tumor suppressor protein, whose loss of function leads the cell to genomic instabilities, i.e., the tumor sets in) [25]. However, definitive evidence of this association has not been proven to date.…”

Awakening the sleeping giant: Epstein–Barr virus reactivation by biological agents

“…During this study, it was observed that the reactivation of EBV occurred and displayed a high load of 3000 genome equivalents per ml (geq/ml), which was further reduced upon treatment with rituximab (Agteresch et al 2007 ). Several studies have shown that tumor generation occurs in Leishmania-infected individuals (Schwing et al 2019 , Ouattassi et al 2022 , Vega-Benedetti et al 2022 ). However, oncogenic EBV involvement remain unexplored in these tumors.…”