ARTICLE
Old World cutaneous leishmaniasis is primarily caused by Leishmania
tropica and Leishmania major. The classical presentation and
course of this disease is the appearance of papules which progress to
ulcers and then spontaneously heal with scarring over months to years.
However, many other clinical forms have been reported and include the
nodular, hyperkeratotic, eczematoid, sporotrichoid, zosteriform, warty,
impetiginized and lupoid forms [1]. Erysipeloid leishmaniasis is yet another
clinical form of cutaneous leishmaniasis which has been reported and is
a rare and unusual presentation. To our knowledge this form of presentation
has previously only been reported from Iran [2, 3].
Case reports
During a clinical trial to treat cutaneous leishmaniasis (CL) patients
with different treatment protocols, 5 patients with an unusual clinical
presentation of CL were observed. All patients were elderly with an age
range between 50 and 70 years of age and predominantly women (4 females
and 1 male). The onset of disease was from 1 to 7 months and the diagnosis
of CL was confirmed by positive skin smears and/or histopathological examinations
(Table I).
On presentation all of the patients had an infiltrative erythematous
plaque covering the center of the face (nose and cheeks) with varying
degrees of scaling and resembling erysipelas (Figs.
1 and 2).
No lymphadenopathy or mucous membrane involvement was detected and general
physical examination was otherwise unremarkable. According to the patients'
histories, the lesions began as diffuse red papules over the center of
the face which slowly coalesced to form a red infiltrative plaque. An
interesting point that was observed in two of the patients was an ulcerated
inoculation site on one cheek which appeared to be the focal point for
the spread of the lesion. In two of the patients the degree of scaling
was much more pronounced and the lesions appeared wet and impetiginized.
The male patient had pock scars over the nose and cheeks due to an old
smallpox infection and the erythematous infiltrative plaque covered much
of the same area.
Routine laboratory examinations were normal.
In four of the patients the skin smears were positive for CL and Leishman
bodies were observed (Fig. 3),
while histopathological examination of a skin biopsy from the case with
a negative smear was diagnostic for CL. In one patient despite a positive
skin smear, a skin biopsy was also performed at another center which was
also diagnostic for CL. All of the patients were then treated by a topical
herbal extract and they were all cured 6 weeks post-treatment with no
scar formation.
Discussion
Cutaneous leishmaniasis (CL) is a spectral disease. Clinically the manifestations
of Old World CL varies from a single papule or nodule which heals within
a few months to chronic or lupoid forms with a duration of over 10 years
[4, 5]. No direct correlation has been reported as to age, sex and clinical
features of CL except for the erysipeloid type of CL which seems to predominantly
affect middle-aged and elderly females. Erysipeloid leishmaniasis, which
to our knowledge has only been reported from Iran, appears to be an unusual
clinical presentation of CL. In a previous clinical study of CL in the
city of Isfahan, Iran, in which a total number of 1,250 patients were
studied, only 17 patients (1.36%) were of the erysipeloid type [3].
Sixteen out of 17 patients were females aged 50-75 years. Thirteen patients
had a disease of less than one year's duration while the other four had
the disease for over one year. All of the patients had presented with
a diffuse erythematous lesion over the face and nose without ulceration
and they all had responded to treatment without leaving any scars. The
clinical findings in the study from Isfahan is very similar to our study.
The reasons for such an unusual clinical presentation
are not clear. However, factors such as skin fragility and friability
of the face especially over the cheeks and nose due to senility, the hormonal
changes in females at or around menopause, and the decrease in host immune
mechanisms with ageing should be considered. It would be of interest to
study the HLA of these patients in order to see if this unusual form of
CL could be at least partly due to host determined factors. Another important
point to consider is that a specific strain of Leishmania could
act as the causative organism. In a recent study in Iran, L. infantum
was reported, for the first time, to be the causative organism in two
cases of cutaneous leishmaniasis [6]. Therefore, it seems prudent to identify
the specific strain(s) involved in cases of erysipeloid leishmaniasis
in order to better understand this peculiar presentation. An interesting
point regarding the role of the organism in this type of presentation
is the possibility of the existence of several different populations of
Leishmania promastigotes within the vector sandflies. One of these
populations may become the predominant population under certain pressures
from the host (i.e. age, sex, hormonal changes, immune status,
etc.) and acts as the causative organism in these cases.
Although all of the above factors are speculations which need further
investigation for confirmation, it seems likely that the pathogenesis
involved in this unusual presentation will be multifactorial.
REFERENCES
1. Dowlati Y. Cutaneous leishmaniasis: clinical aspect. Clin Dermatol
1996; 14: 425-32.
2. Momeni AZ, Amindjavaheri M, Ghanadi F. Erysipeloid type of leishmaniasis:
a clinical study of an unusual presentation of cutaneous leishmaniasis
in Iranian women in Isfahan, Iran. Med J Islam Rep Iran 1994; 7:
273-6.
3. Momeni AZ, Aminjavaheri M. Clinical picture of cutaneous leishmaniasis
in Isfahan, Iran. Int J Dermatol 1994; 33: 260-5.
4. Petit JHS. Chronic (lupoid) leishmaniasis. Br J Dermatol 1962;
74: 127-31.
5. Ardehali S, Sodefy M, Haghighi P, Rezai H, Vollum D. Studies on chronic
lupoid leishmaniasis. Ann Trop Med Para 1980; 74: 439-45.
6. Hatam GR, Hosseini SMH, Ardehali S. Dermotropic isolates of Leishmania
infantum in Iran. Trans Roy Soc Trop Med Hyg 1997; 91: 440.
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