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American Journal of Dermatopathology:
doi: 10.1097/DAD.0b013e31820977d1
Letters to the Editor

Cutaneous Leishmaniasis With Pseudoepitheliomatous Hyperplasia Simulating Squamous Cell Carcinoma

Quintella, Leonardo Pereira MD; Cuzzi, Tullia PhD; de Fátima Madeira, Maria PhD; Valete-Rosalino, Cláudia Maria PhD; de Matos Salgueiro, Mariza MD; de Camargo Ferreira e Vasconcellos, Érica MD; Mouta-Confort, Eliame PhD; Lambert Passos, Sonia Regina PhD; de Oliveira Schubach, Armando PhD

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Instituto de Pesquisa Clínica Evandro Chagas (IPEC), Fundação Oswaldo Cruz (FIOCRUZ), Rio de Janeiro, Brasil

This work was partially funded by Conselho Nacional de Desenvolvimento Científico e Tecnológico (CNPq), Programa Estratégico de Apoio à Pesquisa em Saúde (PAPES-CNPq/FIOCRUZ), and Fundação de Amparo à Pesquisa do Estado do Rio de Janeiro (FAPERJ).

The authors declare no conflict of interests.

To the Editors:

In Rio de Janeiro state, Brazil, American tegumentary leishmaniasis is caused by Leishmania (Viannia) braziliensis. The main clinical presentation is a single skin ulcer located in areas exposed to vector bites. Diagnosis is established by isolation of Leishmania in culture or by amastigote detection in smears or histological sections, but parasitological demonstration is not always attained.1 Histopathological findings include granulomatous dermatitis and occasionally pseudoepitheliomatous squamous hyperplasia (PESH).2 PESH is considered a response to chronic epithelial irritation of various etiologies and can simulate squamous cell carcinoma (SCC).3,4 Cutaneous neoplasms are also frequently located on exposed areas of the body and are important in leishmaniasis differential diagnosis.5

A 59-year-old man from an endemic leishmaniasis rural area in Rio de Janeiro presented with swelling and redness of the left ear and a nodule on the right forearm, both evolving into ulceration in 3 months. Physical examination disclosed ulcers with raised firm edges on the right forearm (Fig. 1A) and lower pendular portion of the left ear lobe (Fig. 1B), a palpable lymph node beneath the angle of the jaw, and no lesions on mucosae of the upper aerodigestive tract.

Figure 1
Figure 1
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Leishmanin skin test was positive, with 21 mm of induration. Indirect immunofluorescence and enzyme-linked immunosorbent assay for leishmaniasis were negative on admission. Biopsy tissue sample submitted to histological examination measured 9 × 4 mm and was 3 mm thick. On microscopy, there was a squamous epithelial downgrowing proliferation with irregular contours and infiltrating aspect (Fig. 1C), arranged in cords and small nests (Fig. 1D) or occasionally, in concentric disposition, forming horny pearls (Fig. 1E). Some cords and nests were situated deep in the dermis. Cytological atypia or atypical mitoses were absent. There was mild mixed inflammatory infiltrate (Fig. 1C) and no granulomas. Two pathologists examined the slides and reported “atypical squamous proliferation consistent with well-differentiated SCC,” with observations that the lesion was partially resected, the sample was superficial, and a hyperplastic process could not be ruled out. Parasitological culture in Novy-Nicolle-McNeal medium and subsequent isoenzyme analysis identified L. (V.) braziliensis. Histological slide review revealed rare structures consistent with Leishmania sp. amastigotes (Fig 1E, insert).

Treatment was started with meglumine antimoniate for 30 days. Three months after the end of therapy, lesions were reepithelialized. Histopathology of a new biopsy sample of the arm lesion showed fibrosis and very focal small granulomas. No epithelial proliferation or amastigotes were seen, and the lesion was considered consistent with a healing process. Patient was lost to follow-up.

PESH is a microscopic pattern of tissue reaction characterized by uneven growth of squamous cells toward underlying connective tissue. It can be distinguished from SCC by the coexistence of an exuberant inflammatory infiltrate and the lack of histological and cytological signs of malignancy.6 Grunwald et al3 stated that horny pearls are more frequent in SCC but can be seen in PESH.

Close clinicopathological correlation beside good communication between attending physicians and pathologists can avoid mistaken diagnoses and unnecessary therapeutic procedures.7,8 Our case also emphasizes the importance of generous sampling and multidisciplinary approach in the differential diagnosis of cutaneous ulcers. A diagnosis of SCC may also be missed if histopathological examination is not included in this investigation.9

Concomitance of American tegumentary leishmaniasis and cutaneous neoplasms is theoretically possible and was recently reported,10 but the patient's evolution and findings on second biopsy make this possibility extremely unlikely.

Leonardo Pereira Quintella, MD

Tullia Cuzzi, PhD

Maria de Fátima Madeira, PhD

Cláudia Maria Valete-Rosalino, PhD

Mariza de Matos Salgueiro, MD

Érica de Camargo Ferreira e Vasconcellos, MD

Eliame Mouta-Confort, PhD

Sonia Regina Lambert Passos, PhD

Armando de Oliveira Schubach, PhD

Instituto de Pesquisa Clínica Evandro Chagas (IPEC), Fundação Oswaldo Cruz (FIOCRUZ), Rio de Janeiro, Brasil

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REFERENCES

1. de Oliveira-Neto MP, Mattos MS, Perez MA, et al. American tegumentary leishmaniasis (ATL) in Rio de Janeiro State, Brazil: main clinical and epidemiologic characteristics. Int J Dermatol. 2000;39:506-514.

2. Dimier-David L, Ravisse P, Bustillos R, et al. [Histopathology of mucocutaneous leishmaniasis caused by Leishmania (Viannia) braziliensis]. Ann Dermatol Venereol. 1994;121:387-392.

3. Grunwald MH, Lee JY, Ackerman AB. Pseudocarcinomatous hyperplasia. Am J Dermatopathol. 1988;10:95-103.

4. Hurwitz RM. Pseudocarcinomatous or infundibular hyperplasia. Am J Dermatopathol. 1989;11:189-192.

5. Grevelink SA, Lerner EA. Leishmaniasis. J Am Acad Dermatol. 1996;34(2 pt 1):257-272.

6. Murphy GF, Elder DE. Non-melanocytic tumors of the skin. In: Rosai J and Sobin LH, ed. Atlas of tumor pathology. Washington, DC: Armed Forces Institute of Pathology; 1991. Available from the American Registry of Pathology.

7. Khorsandi-Ashtiani MT, Hasibi M, Yazdani N, et al. Auricular leishmaniasis mimicking squamous cell carcinoma. J Laryngol Otol. 2009;123:915-918.

8. Gacek MR, Gacek RR, Gantz B, et al. Pseudoepitheliomatous hyperplasia versus squamous cell carcinoma of the external auditory canal. Laryngoscope. 1998;108(4 pt 1):620-623.

9. Fagundes A, Schubach A, de Paula CC, et al. Evaluation of polymerase chain reaction in the routine diagnosis for tegumentary leishmaniasis in a referral centre. Mem Inst Oswaldo Cruz. 2010;105:109-112.

10. Karabekmez FE, Duymaz A, Keskin M, et al. Squamous cell carcinoma on cutaneous leishmaniasis lesion. Dermatol Surg. 2008;34:1742-1743.

© 2011 Lippincott Williams & Wilkins, Inc.

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