Association between periorbital cutaneous leishmaniasis and basosquamous carcinoma

A 73-year-old male rural worker from the Brazilian Amazon region presented with a 4-year left facial ulceration, which became painful as it increased in size. Physical examination revealed a deep 8-cm ulcer in the left malar and periorbital regions, associated with necrosis and purulent exudation (Figure 1). Skull computed tomography showed an osteolytic and infiltrative lesion in the malar and frontal regions (Figure 2).

FIGURE 1:
Ulcerated lesion with necrosis and bone exposure.

FIGURE 2:
Destruction of the left orbit and left zygomatic bone. Left maxillary sinus filling (skull computed tomography).

Histopathological examination of the lesion revealed atypical basaloid and squamous cell proliferation, with ovoid microstructures within the histiocyte cytoplasm, suggestive of leishmaniasis (Figure 3). Polymerase chain reaction identified Leishmania viannia. Immunohistochemical analysis of the samples revealed P-63-positivity, whereas the epithelial membrane was antigen-negative, confirming the diagnosis of basosquamous carcinoma (BSC).

FIGURE 3:
Image of skin sample showing basaloid cells with scarce cytoplasm and hyperchromatic, irregular nuclei (blue circles); mitotic figure (blue arrow); lympho-histio-plasmocitary inflammatory infiltrate (red circle); vacuoles with ovoid corpuscles within histiocytes (red arrows) (haematoxylin & eosin staining; 400x magnification).

Treatment with meglumine antimoniate for tegumentary leishmaniasis (TL) produced a satisfactory clinical response. The patient underwent radiotherapy and enucleation and was scheduled for reconstruction.

To our knowledge, this is the first case describing the association between BSC and TL. TL may impair the immune system, thereby reducing clonal cell susceptibility to destruction¹. This case highlights leishmaniasis as a differential diagnosis of complicated mucocutaneous lesions², thus establishing a new reference for future studies investigating the interaction between infectious and neoplastic diseases, and it may even strengthen recent hypotheses of a direct correlation between leishmaniasis and skin carcinomas³.

REFERENCES

• ¹
  Kopterides P, Mourtzoukou EG, Skopelitis E, Tsavaris N, Falagas ME. Aspects of the association between leishmaniasis and malignant disorders. Trans R Soc Trop Med Hyg. 2007;101(12):1181-9.
• ²
  Tarallo M, Cigna E, Frati R, Delfino S, Innocenzi D, Fama U, et al. Metatypical basal cell carcinoma: a clinical review. J Exp Clin Cancer Res. 2008;27(1):65.
• ³
  Bogdan C. Leishmaniasis in rheumatology, haematology and oncology: epidemiological, immunological and clinical aspects and caveats. Ann Rheum Dis. 2012; 71(Supp II): i60-6.

Publication Dates

• Publication in this collection
  08 Mar 2021
• Date of issue
  2021

History

• Received
  21 Oct 2020
• Accepted
  25 Nov 2020