Unusual Form of Cutaneous Leishmaniasis: Erysipeloid Form

European Annals of Otorhinolaryngology, Head and Neck diseases (2011) 128, 95—97

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CLINICAL COMMENTARY Unusual form of cutaneous leishmaniasis: Erysipeloid form

M. Mnejja a, B. Hammami a, A. Chakroun a, I. Achour a, I. Charfeddine b, A. Chakroun a, H. Turki a, A. Ghorbel a,∗

a Service ORL et chirurgie cervico-faciale, CHU Habib Bourguiba, 3029 Sfax, Tunisia b Service de dermatologie, CHU Hedi Chaker, Sfax, Tunisia

Available online 19 January 2011

KEYWORDS Summary We report the epidemiological and clinical characteristics of the erysipeloid form Leishmaniasis; of cutaneous leishmaniasis as well as its diagnostic and therapeutic challenges. Cutaneous; Case report: A 63-year-old woman, with no medical history, presented with a one-month history Erysipeloid of erythematous nasal swelling. The lesion appeared after an accidental trauma. Erythematous infiltrative plaque was noted on the center of the face. There were also crust formations on the traumatic region. Despite local treatment and oral antibiotherapy, there was no improvement. The diagnosis of cutaneous leishmaniasis was confirmed by positive skin smears. Histopathologi- cal examinations of a skin biopsy showed no malignancy. The patient was treated intramuscularly with 10 mg/kg per day systemic meglumine antimoniate with partial regression of symptoms. Conclusion: The erysipeloid type is a rare and unusual presentation of cutaneous leishmaniasis that often causes late diagnosis. Diagnosis is confirmed by the demonstration of the parasite by skin smear, histopathological examination and polymerase chain reaction. There are various therapeutic options. The evolution is generally favourable. © 2011 Elsevier Masson SAS. All rights reserved.

Introduction quent parasite encountered in Southern parts of the country [1]. Cutaneous leihmaniasis is characterized by clinical poly- Cutaneous leishmaniasis (CL) is a world widespread parasitic morphism. Erysipeloid form is a rare and unusual entity. disease caused by the infectious bite of an insect vector, the We report a case of erysipeloid form of CL of the phlebotomine sandﬂy. In Tunisia, it has become a very com- face in order to examine the epidemiological, clinical and mon disease for several years, with an endemo-epidemic therapeutical features of this form. mode of evolution. Leishmania major is by far the most fre- Observation

∗ Corresponding author. Tel.: +216 74 243 979; A 63-year-old female patient had no particular past-medical fax: +216 74 243 979. history. She consulted for a one-month inﬂammatory nasal E-mail address: [email protected] (A. Ghorbel). swelling that occurred after nasal trauma. Pristinamycine

temic meglumine antimoniate (Glucantime®) administered intramuscularly was conducted. The evolution was marked by partial healing of the lesions.

Discussion

In Tunisia, the classic form of CL is nodular with ulcers and crusts. The manifestation is an infiltrative nodule with a central crater covered with a yellow brownish crust. Spon- taneous healing occurs within a few months with filling of ulcer, leaving a clear or pigmented scar and conferring lasting immunity [1]. Cases of CL in its erysipeloid form have been reported in Iran, Pakistan, Turkey and Tunisia [1—4]. It differs from the other forms by its clinical features but also by the predominantly affected population [4]. In the literature, the incidence rate of erysipeloid form of CL ranges between 0,05 and 3,2% [1,3]. This type pre- Figure 1 Erythemateous infiltrative plaque asymmetrically dominantly affects elderly females [4]. Clinical features are covering the center of the face. erythematous infiltrative ill-defined plaque over the face covering the nose and both cheeks [1]. was administered orally for 7 days without clinical improve- The etiology of this type is unknown. Altered host ment. Physical examination revealed an erythematous immune response due to senility, a specific type of parasite, infiltrative plaque covering the center of the face (nose and hormonal changes at menopause, skin quality alteration cheeks) with a grossly symmetrical pattern of 5 cm lesions due to ageing were evoked to explain this particular form covered by places with crusts (Fig. 1). Endonasal exami- [1,2,4]. Post-traumatic cutaneous lesions can facilitate the nation did not reveal any mucosal lesion and the rest of occurrence of this type of disease [5]. the examination was normal. The patient was apyretic and Our patient presented with all the epidemiological fea- the biological and inflammatory evaluation tests were nor- tures characterizing this particular form. She was native of mal. The patient was admitted to hospital and received an endemic region of leishmaniasis, of old age, menopaused broad spectrum antibiotic treatment with daily local care with a history of nasal trauma followed by the occurrence in prevention of post-traumatic cellulitis risk. Evolution of the nasal lesion. was marked by the absence of clinical improvement with In cases of localized form, these lesions can evoke bac- a tendency to spreading of erythematous lesions (Fig. 2). terial or fungal skin infection, syphilis, anthrax, eczema, Biopsy of the lesion did not reveal any sign of malignancy. tuberculosis, infected insect bite or primary or metastatic At that stage, CL was suspected and confirmed by posi- skin tumor. In cases of facial lesions, differential diag- tive nasal tissue smears, which demonstrated leishmania nosis must be made with disseminated or discoid lupus amastigotes forms. A treatment with 10 mg/kg per day sys- erythematous, lupus vulgaris, sarcoidosis or erysipelas [4,6]. In Tunisia, CL diagnosis is based on direct parasitology tests to detect leishmania parasites, DNA research of leih- maniases via PCR (a new technique recently used in Tunisia) and on the histopathological examination of skin biopsy [7]. There are several therapeutic options such as cryother- apy, heat therapy with radiofrequency, topical treatment, oral treatments such as fluconazole, metronidazole [8].In Tunisia, the pentavalent antimony (meflumine antimoniate) remains the treatment mainstay. Parenteral antimony administration is used with a 10 mg/kg per day dosage twice daily for 20 days [6]. Parenteral administration is recom- mended in cases of multiple or severe lesions or in lesions causing severe cosmetic sequelae on the face, for exam- ple. Erysipeloid form involving the face requires injectable treatment as in the case of our patient. A treatment with topical herbal extract (Z-HE) was described by Zerehsaz and al [9] in this particular form of disease with a healing rate without recurrence of 92% after 12-month follow-up. Low cost, easy preparation and few drug related side effects characterize this topical treat- Figure 2 Erysipeloid cutaneous leishmaniasis of the face: ment but further studies are necessary in order to confirm spreading of the lesions after antibiotic treatment. its efficiency. Unusual form of cutaneous leishmaniasis: Erysipeloid form 97

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