Diffuse cutaneous leishmaniasis due to Leishmania major in Senegal

TRANSACTIONSOFTHEROYALSOCIETYOFTROPICALMEDICINEANDHYGIENE(1996)90,396-397

maniasis: L. major, (see VENENCIE et al., 1993),L. braziliensis (see COURA et al., 1987), and L. infantum (see JIMENEZ et al., 1991).

IShort ReDortl I L Diffuse cutaneous leishmaniasis to Leishmania major in Senegal

due

Michel Develouxl, Samba Diallo*, YCmou Dieng*, Ibrahima Mane3, Michel Huerre4, Francine Pratlongs, Jean-Pierre Dedets* and Bassirou Ndiayel ‘Unique de Dermatologie, CHU Hopital A. Le Dantec, BP. 3001, Dakar, Senegal; ‘Service de Parasitologic, Faculte’ de Medecine et Pharmacie, Dakar, Senegal; 31nstitut de Ltiprologie, Centre Hospitalier de Farm, Dakar, Senegal; 4Unite’ d’Hi.stopathologie, Institut Pasteur de Paris, Paris, France; 5Laboratoire d’Ecologie Medicale et Pathologie Parasitaire, Faculte’ de Medecine de l’liniversite de Montpelher, Montpellier, France Keywords: diffuse cutaneous leishmaniasis, Leishmania major, Senegal

Diffuse cutaneous leishmaniasis (DCL) is an unusual form of cutaneous leishmaniasis generally resulting from the infection of anergic patients by Leishmania aethiopica in the ‘Old World’ or L. amaaonensisin the ‘New World’.

We report in the present paper a caseof DCL due to

L. major in Senegal. Case report

A 57 years old woman from Koungheul, Kaolack district, Senegal,was referred to Dakar with a presumptive diagnosis of lepromatous leprosy. Papular lesions had appearedone year earlier on her forehead. Over the next few months, she developed nodules on her face and then over her entire skin surface. On examination she had infiltrations and nodules on the face (nose, eyebrows, ears), giving her a ‘leonine’ appearance (Fig. 1). Numerous nodules and plaques were also present on her trunk (Fig. l), arms and legs. The nodules were firm and copper-coloured. No ulcerated lesion and no mucosal involvement were found. Peripheral nerves were normal and skin sensation was intact. No hepatosplenomegaly or adenopathy were detected. Her clinical condition and nutritional state were otherwise good. Leprosy bacilli were not found, but Giemsa-stained slit-skin smears of nodular lesions showed numerous amastigotes of Leishmania. Culture in NNN medium resulted in the isolation of Leishmania (strain MHOMISNI93ISAMDIA). Starch gel electrophoresis,

Fig. 1. Patient with diffuse cutaneous leishmaniasis causedby L. major. Nodules on the face, giving ‘leonine’ appearance (left), and on the back (right).

The first casesof DCL were reported from Venezuela by CONVIT & LAPENTA (1948) and from Ethiopia by DESTOMBES et al. (1965). Further sporadic cases have been regularly reported from Ethiopia, Kenya, various countries in South America (DEDET, 1993), and Mexico (VELASCO et al., 1989). Since cases of acquired immunosuppression, either therapeutically induced or related to infection with human immunodeficiency virus (HIV), are increasing, a few DCL caseshave been reported related to other Lezshmania species, which are usually responsible for localized cutaneous leishmaniasis or mucocutaneous leish*Author for correspondence.

using 15 enzymes according to the technique of RIOUX et al. (1990), led to the identification of the isolates as L. major, zymodeme MON-26. Histological examination of a nodule biopsy showed that the epidermis was intact but thin and flat, without hyperkeratosis. Under the epidermis, there was a thin clear zone; the rest of the dermis was obliterated by a massive infiltration consisting of macrophagesand lymphocytes. The macrophages were vacuolated or epithe-

lioid with numerous amastigotes in their cytoplasm (Fig. 2). Blood cell count revealed lymphopenia: 1024x106/L, with a CD4+/CD8+ ratio of 0.6. No other biological ab-

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pressed (VENENCIE et aZ., 1993). In our case, the immunological status of the patient was not known. Despite lymphopenia and the sudden death of the patient, HIV-l and 2 and HTLV-I serologies were negative. Acknowledgements

We thank MS M. Lefebvre and Mr P. Lami for expert technical assistance,and Dr R. N. Davidson for revising the manuscript. References

Abdel-Hameed, A. A., Ahmed, B. O., Mohamedani, A. A., ElHarith, A. & Van Eys, G. (1990). A case of diffuse cutaneous leishmaniasis due to Leishmania major. Transactions of the Royal Society of Tropical Medicine and Hygiene, 84, 535-536.

Bryceson, A. D. M. (1969). Diffuse cutaneous leishmaniasis in Ethiopia. I. The clinical and histological features of the disease. Transactionsof the Royal Society of Tropical Medicine and Hygiene, 63,708-737. Convit, J. & Lapenta, P. (1948). Sobre un case de leishmaniasis tegumentaria de forma diseminada. Revista de la l’oliclinica, Caracas, 17,153-158.

Fig. 2. Section of skin from patient with diffuse cutaneous leishmaniasis, showing epidermal and dermal infiltrate containing a majority of vacuolated histiocytes containing numerous amastigotes of L. major (safranin-eosin-haemalum stain, x 1000). normality was found. Serological tests for HIV-l and 2 and human T lymphotrophic virus type I (HTLV-I) were negative. High titres of antileishmanial antibodies were detected: l/3200 in a fluorescent antibody test using L. donovani antigen. Counter electrophoresis was positive, with 3 precipitin lines. The leishmanin skin test was not performed. The patient was treated with meglumine antimoniate (Glucantime@) at a dosage of 17 mg Sb/kg/d for 15 d. Marked regression of the lesions was observed. She received a second course of meglumine antimoniate one month later. Post-treatment smears showed scanty amastigotes. The patient died of an unknown cause 6 months later in her village.

Coura, J. R., Galvao-Castro, B. & Grimaldi, G., jr (1987). Disseminated American cutaneous leishmaniasis in a patient with AIDS. Memorias do Instituto Oswald0 Cruz, 82, 581-582. Dedet, J.-P. (1993). Leishmania et leishmanioses du continent americain. Annales de l’lnstitut Pastuer, Actualit& 4, 3-25. Dedet, J.-P., Marchand, J. P, Strobe& M. & Derouin, F. (1979). Ecologic d’un foyer de leishmaniose cutanee dans la region de ThiPs (Senegal, Afrique de 1’Ouest). 2.-Particularites Cpidemiologiques et cliniques de la maladie humaine. Bulletin de la Sock% de Pathologie Exotique, 72,245-253.

Dedet, J.-P., Safjanova, V. M., Desjeux, P., Emelyanova, L. P., Schnur, L. P. & Chance, M. L. (1982). Ecologic dun foyer de leishmaniose cutanee dans la region de Thies (Senegal, Afrique de I’Ouest). VI. Caractirisation et typage des souches de Leishmania isolees.Bulletin de la Socit% de Pathologie Exotique, 75,155-168. Dedet, J.-P., Safjanova, J. M., Le Blancq, S. M., Desjeux, P., Schnur, L. F., Emelyanova, L. l’. & Chance, M. L. (1986). Caracterisation de souches de Leishmania major Yakimoff et Schokhor, 1914, isolees au Senegal (Afrique de 1’Ouest).Jn: Leishmania: Taxonomie et Phylogenbe. Applications Ecoipidimiologiques, Rioux, J. A. (editor). IMEEE, Montpellier, pp.463-469. Desjeux, P., Waroquy, L. & Dedet, J.-P. (1981). La leishmaniose cutanee humaine en Afrique de l’Ouest. Bulletin de la Soci& de Pathologie Exotique, 74,414-425.

Discussion

Senegal is located in the Sahelien belt, where cutaneous leishmaniasis due to L. major is endemic (DESJEUX et al., 1981; DEDET et al., 1982) and is caused by zymodeme MON-26 (=LON-1) (DEDET et al., 1986). The disease consists of localized cutaneous lesions, generally of the ulcerative type (DEDET et al., 1979). To our knowledge, our patient is the first case of DCL reported from West Africa. The clinical presentation was similar to that of the Ethiopian cases, simulating lepromatous leprosy (BRYCESON, 1969). Involvement of the trunk, rarely observed in Ethiopian DCL patients, was present in this case. DCL due to L. major has previously been reported twice: first, in a Sudanese male with pre-existing lymphoedema as a probable predisposing factor in the pathogenesis of his condition (ABDEL-HAMEED et al., 1990), and second in an Algerian woman who was a kidney transplant recipient and was therefore immunosup-

Destombes, P., Poirer, A. & Serie, C. (1965). Leishmaniose cutanee nodulaire disseminee en Ethiopie. Archives de l’lnstitut Pasteur d’Rlgt+ie, 43,9-19.

Jiminez, M. I., Guttierez-Solar, B., Benito, A., Aguiar, A., Garcia, E., Cercenado, E. & Alvar, J. (1991). Cutaneous Leishmania (L.) infantum zymodemes isolated from bone marrow in AIDS patients. Research and Reviews in Parasitology, 51,95-99. Rioux, J. A., Lanotte, G., Serres, E., Pratlong, F., Bastien, P. & P&i&es, J. (1990). Taxonomy of Leishmania. Use of isoenzymes. Suggestionsfor a new classification. Annales de Parasitologie Humaine et Comparde, 65, 111-125. Velasco, O., Savarino, S. J., Walton, B. C., Gam, A. A., & Neva, F. A. (1989). Diffuse cutaneous leishmaniasis in Mexico. American Journal

of Tropical

Medicine

and Hygiene,

41,

280-288. Venencie, P. Y., Bouree, P., Hiesse, C., Viellefond, A., Lantz, O., Doukan, S. & Rioux, J, A. (1993). Leishmamose cutanee disseminee chez une immunodeprimee. Annales de Dermatologie et de V&tblogie, Received 12 April

120,461-463.

1996; accepted for publication

1 May 1996