Unusual multifocal cutaneous leishmaniasis in a diabetic patient

ARTICLE

Auteur(s) : Ali Murat Ceyhan, Mehmet Yildirim, Pinar Yuksel Basak, Vahide Baysal Akkaya

Suleyman Demirel University, Faculty of Medicine, Department of Dermatology, 32100 Isparta, Turkey

A 60-year-old woman presented with multifocal facial lesions on the right side of the nose and the right forearm. According to the patient, the initial lesion started as a small insect-bite-like red papule at the right mandibular region eight months previously, spreading to involve the right side of the cheek, nose and right forearm. She had no subjective symptoms, including pruritus, fever or pain. Prior treatment with oral/topical antibiotics or topical steroids was ineffective. Medical history included hyperlipidemia, hypertension and a 15-year history of type 2 diabetes mellitus requiring oral antidiabetics. Physical examination showed an infiltrated erythematous plaque of about 5 cm in diameter on the right cheek in association with painless erythematous papular, nodular and tumoral lesions covered with a yellowish crust spreading to the right medial epicantus, upper nose, mandibular ramus and upper side of the chin (figure 1A). On the extensor right forearm, there were multiple erythematous nodular lesions, varying from 10 mm to 15 mm in diameter with superficial central ulcers, and an eczematous plaque was located on the dorsum of the right hand (figure 1B). A total of 14 lesions were observed.

Results of routine laboratory tests were all within normal limits except for hyperglycemia and mild anemia. Serotesting for HIV was negative. Histopathological examination of the facial erythematous plaque showed numerous amastigotes with a dense mixed inflammatory infiltrate in the dermis. A Giemsa-staining skin smear of papulonodular and eczematoid lesions revealed numerous amastigotes. Leishmania tropica DNA was detected in the facial erythematous lesion using PCR technology. The patient was successfully treated with meglumine antimonate (Glucantime^®), 15 mg/kg intramuscularly daily for 20 days.

Our patient exhibited some atypical characteristics, i.e. the multifocal distribution and a distinct, unusual morphological pattern of the lesions. Aghaei et al. reported a case of ulcerated disseminated CL associated with diabetes mellitus in a patient with Down syndrome [1]. To the best of our knowledge, there is no report yet of multifocal CL associated with diabetes mellitus. Unusual forms of CL such as erysipeloid, squamous cell carcinoma-like variant and eczematoid type have been reported in individual patients [2, 3]. However, the coexistence these types of CL in the same patient, as described here, has not been documented in the literature.

The different clinical pictures of CL depend on multiple factors, such as Leishmania subspecies, number of inoculated parasites, site of inoculation, length of infection, steroid usage, HIV infection, and cellular immunity [3]. The immunological response to leishmaniasis is complex and largely T-cell-mediated, so that early T cell and cytokine responses partially determine the outcome of infection. In Leishmania infections, the ability of host macrophages to effectively kill the intracellular parasite limits the disease, which usually requires the production of adequate IFN-γ by Th1 cells to activate infected macrophages [4]. Diabetes mellitus has long been considered as an important risk factor for infection, and is indeed recognized by the World Health Organization as a cause of secondary immunodeficiency [5]. The defective host immune factors caused by diabetes mellitus in our patient may elicit a poor T-cell response to Leishmania antigens leading to these unusual characteristic features. Several studies have demonstrated the inhibitory effects of steroids on cellular immune responses. Indeed, pre-exposure of CD4+ lymphocytes to steroids suppressed the secretion of Th1-type cytokines, such as interferon (IFN)-γ and tumor necrosis factor (TNF)-α [6].

In our case, multiple sand fly bites and increasing skin fragility due to senility may have caused the unusual morphology and multifocal distribution of CL. It may also be explained by the defective host immune factors caused by diabetes mellitus and the previous inappropriate use of steroids.

Although determinants of the unusual different clinical presentations of CL remain poorly understood, an inadequate cell-mediated immunity to Leishmania antigen probably leads to uncontrolled parasite growth. The coexistence of diabetes mellitus should be taken into account in such cases.

Acknowledgements

References

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