Mucocutaneous leishmaniasis in an 11-year-old girl with ataxia telangectasia – case report

نویسندگان

  • Safa S. Elfaituri
  • Idris Matoug
  • Hanan Elsalheen
  • Yousif Belrasali
  • Fatma Emaetig
چکیده

Mucocutaneous leishmaniasis in an 11-year-old girl with ataxia telangectasia Á case report L eishmaniasis is a protozoal disease caused by flagellates of the genus Leishmania and transmitted by sand fly. The reservoir hosts are humans, dogs, and rodents. It has three different morphological forms: cutaneous, mucocutaneous (MCL), and visceral. The different clinical presentations depend on the interaction between parasite species of leishmania and the genetic and immunological status of the host (1). An 11-year-old girl, a known case of ataxia telangectasia, from Ghaser Libya near Wadi Alkof presented with complaints of nasal skin lesion of 5 months duration, followed by purulent rhinitis and destruction of anterior nose with involvement of adjacent area of skin and upper lip. Clinically, telangectasia was evident in conjunctivae (Fig. 1) and skin of face. On walking she had ataxic gait. She had an ulcerated lesion on the anterior part of nose with central purulent crust and raised border. Erythema-tous papulopustular lesions and superficial small ulcers were seen around the nose, cheeks, upper lip (Fig. 2), and hard palate. Nasal examination revealed congested septum, mucopus in nasal cavity, and nasal ulceration with cartilage destruction. Serology for HIV was negative. Amastigotes inside macrophage were seen by Leishman's stained scraping smears (Fig. 3) and by Giemsa stained histopathological section (Fig. 4). Treatment with sodium stibogluconate 20 mg/kg/day was administered for 28 days with amphotericin 1 mg/kg. Improvement was only partial, and the patient relapsed once treatment discontinued. Discussion Diagnosis of leishmaniasis is based on criteria that consider epidemiological data, clinical features, and laboratory test results: direct smear, culture and inoculation , biopsy, and PCR (2). Based on the clinical picture, the immune state of our patient and the geographical area she came from Á Ghaser Libya from where many cases of cutaneous leishmaniasis had been reported Á a clinicoepidemiological link was made and MCL was considered as a possible diagnosis, and it was confirmed by demonstrating the leishmania parasites. MCL is considered to be a disease of the New World; it is almost exclusively restricted to South America and is caused typically by Leishmania braziliensis. In rare cases, it may be caused by Old World species like L. aethiopica, L. major, and L. tropica (3, 4). MCL due to L. infantum had been reported in Mediterra-nean countries, largely resulting from HIV co-infection (3). Unfortunately, leishmania species identification was not possible in our patient as it was not available. …

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عنوان ژورنال:

دوره 10  شماره 

صفحات  -

تاریخ انتشار 2015