Cutaneous leishmaniasis in two siblings

ARTICLE

Auteur(s) : Lina Houssami¹, Fadi Haddad¹, Hassan El Teraifi², Thomas G. Berger¹

¹ Division of Dermatology, Tawam Hospital, P.O. Box 15258, Al Ain, Abu Dhabi, United Arab Emirates, Thomas Georg Berger,
<bergerts@web.de>
² Department of Histopathology, Tawam Hospital, Al Ain, Abu Dhabi, UAE

Treatment of cutaneous leishmaniasis in children is a difficult task, since available options are often toxic or painful. Here we report on two siblings with an exceptional clinical response to topical treatment with 20% paromomycin, without scar formation. In addition, we critically discuss the possibility of direct contact transmission between these two children.
Cutaneous leishmaniasis (CL) is endemic in many countries in the Middle East [1]. In the United Arab Emirates the epidemiological situation is unclear, but cases are seen in patients returning from neighbouring countries such as Saudi Arabia and Iran, where CL is a major health problem [2] CL is usually acquired through the bite of an infected sandfly. In addition, CL has been transmitted by deliberate scarification [3] and through breast feeding [4]. Direct transmission due to close contact (e.g. within families) is otherwise almost unreported.
We report here on the exceptional response to topical paromomycin, which was easy to use and well tolerated. In addition, due to the latency of the skin eruption in the girl and the clinical history – the girl kissed her brother frequently on the face – the possibility of direct contact transmission was considered.

Case reports

A 2-year-old boy presented with erythematous and crusted nodules on his left cheek and right thigh of 5 months duration (figure 1A). The family spent the summer in Saudi-Arabia, the parents reported multiple insect bites.
Cytology: A smear from a facial lesion showed abundant leishmania amastigotes (figure 1C), electron microscopic examination confirmed the presence of leishmania parasites (figure 1C, insert).
The 7-year-old sister presented with an erythematous crusted swelling on the upper lip, which appeared 4 months later than in her brother (figure 1B). Insect bites were not remembered.
A smear revealed the same cytological appearance with multiple leishmania parasites (figure 1D).
100 mg itraconazole daily was given orally for 6 weeks to both children without improvement. Therefore, topical paromomycin 20% in a 10% urea-containing formulation was applied twice daily for 6 weeks, with excellent healing and no scar formation (figure 1E and F).
Cutaneous leishmaniasis (CL) expresses various clinical patterns from self-healing sores to extensive lesions with severe disfigurement. To date, there is no ideally safe, simple and effective treatment, especially for small children. Systemic or local chemotherapy with pentavalent antimonials is associated with significant toxicity and requires multiple injections. Azole derivatives are an alternative [5], but not always effective. Likewise, many of the topical treatment options such as curettage, cryotherapy, heat application or photodynamic therapy are painful and therefore not feasible for young children. The topical treatment with paromomycin 20% under occlusive dressing has been proven effective in controlled studies [6]. It is a safe and practically painless therapy. In our cases, topical paromomycin over a total of 6 weeks resulted in excellent healing with no scar formation.
The interesting question remains, did the girl acquire the disease directly by frequently kissing her brother, which was reported by the mother and witnessed by the treating physician? CL is usually transmitted through the bite of an infected sandfly. Since deliberate scarification as a form of active immunotherapy has been reported for centuries, direct contact transmission is in principle possible, however, it is rarely reported [3, 4]. In fact, we did not find a single epidemiological study which has addressed whether or not leishmaniasis can be transmitted by direct contact. This problem is admittedly difficult to tackle, since most cases occur in endemic areas and the incubation period has such a great variability that even if direct transmission is considered clinically, a vector borne transmission can hardly be ruled out. As in our case, the skin around the lips often is macerated which would facilitate the entry of the parasite. Also, the specific location and long latency makes it conceivable that direct contact other than an insect bite transmitted the parasites. Clearly, more information is needed, but we see this correspondence as a starting point to stimulate future studies in that direction. n

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