ARTICLE
The protozoon leishmania, which is transmitted by the bite of a sandfly,
can cause three distinct clinical entities: oriental
sore caused by Leishmania tropica; Kala-azar caused by Leishmani donowani
; and espundia caused by Leishmania braziliensis [1].
Oriental sore is the typical cutaneous lesion.
The eyelid is involved in 2.5% of cases. Conjunctiva are rarely affected
and conjunctivitis is usually secondary to secondary infection [1]. Clinical
diagnosis of ocular leishmaniasis is very difficult and may simulate other
more common lesions such as recurrent chalazion and tumors such as basal
cell carcinoma specially its ulcerative form [2, 3]. Here we report a
biopsy proven case of the mucocutaneous form of ocular leishmaniasis in
a young man.
Case report
A 32-year-old physician who was working in the south west of Iran developed
a non-tender reddish nodule with surrounding erythema on the right lower
eyelid. The case was diagnosed as a hordeolum and was treated with antibiotic
eye drops and ointments. No improvement was noticed and gradually a small
ulceration developed accompanied with edema, echyomosis and erythema.
Enlargement of lesion during the next month was noticed. Visual acuity
was 20/20 in both eyes. There was no corneal involvement and the rest
of the ocular examination was within the normal limits. A biopsy was taken
at this time to rule out basal/squamous cell carcinoma. The pathology
examination disclosed a dense inflammatory infiltration of lymphocytes,
histiocytes and plasma cells, and rarely neutrophils. Many of the histiocytes
contained typical Leishmania organisms. Isolated organisms from ruptured
histiocytes were noted in the interstitia. No malignancy was noticed.
The patient was treated with stibogluconate both intra muscular (20 mg/kg
for 2 weeks) and intra-lesional. The ulcer gradually healed leaving a
small scar, but recurrence was noticed 4 months later. Examination at
this time revealed a red nodule with surrounding erythema at the previous
location (Fig. 1). The
patient complained of a burning sensation in the right eye. Conjunctiva
and episclera were red and inflamed (Fig.
1). Slit lamp examination revealed diffuse nodular episcleritis around
the perilimbal area. Episcleritis did not respond to steroid eye drops.
Treatment with stibogluconate both intra-muscular (20 mg/kg) and intra-lesional
for an additional 3 weeks, resulted in the improvement of both eyelid
and conjunctival lesions.
In follow up four months later, inflammation of the eyelid and episclera
had recurred. At this time a biopsy was performed from bulbar conjunctiva
which revealed a Leishmania parasite in the tissue (Fig.
2). Combined therapy was planned with stibogluconate 20 mg/kg per
day and allupurinol (20 mg/kg per day) for one month. Fifteen days after
starting the combined therapy, the patient developed generalized skin
eruptions, assumed to be a reaction to allupurinol which was discontinued.
Stiboglauconate was continued for another month. At the end of therapy
the eyelid lesion, episcleritis and conjunctivitis healed completely (Fig.
3). Follow up after 2 years revealed only a small scar on the eyelid
and a normal globe. No impairment of vision was noticed.
Discussion
Leishmaniasis is among the six diseases of the UNDP/World Bank/WHO's
special program for research and training in tropical diseases, with a
350 million population at risk [4]. Approximately 12 million are affected
and 1.5 to 2 million infected each year [5]. The disease is more common
in developing countries. Despite various control measures, the incidence
of the disease has increased and new endemic areas are seen due to immigration.
The vectors are sand flies of 2 genera, Lutzonyia in the new world and
phlehotomus in the old world and the reservoirs can be humans and animals
[5]. Depending on the species of the leishmaniasis and the host immune
responses, different clinical pictures have been described such as cutaneous,
mucocutaneous and visceral leishmaniasis.
Cutaneous leishmaniasis is endemic and visceral leishmaniasis is sporadic
in different parts of Iran [6]. However the mucocutaneous form is very
rare.
The lid is rarely involved in the cutaneous form of leishmaniasis possibly
due to the movement of the lids preventing the fly-vector from biting
the skin in this region [1]. The conjunctiva and episclera are also rarely
affected in cases of oriental sore and they may be involved by either
contiguous spread from the skin of eyelid or by inoculation of the conjunctiva
by the patient's own fingers [3].
Although, it is very hard to establish a correct diagnosis in the ocular
involvement of leishmaniasis, it is very important to do so because lesions
caused by leishmania infection may simulate other conditions e.g.
chalazion, dacryocystitis and tumors [2, 7, 8]. A definitive diagnosis
of leishmaniasis can be obtained by a biopsy of a lesion and demonstration
of organisms. The oriental sore is a self limiting condition, however,
if eyelid lesions remain untreated, the contiguous spread from the skin
of the eyelid will extend to involve the conjunctiva, episclera and even
the cornea, with development of interstitial keratitis.
Long term complications of ocular leishmaniasis
include lid deformity, with all its consequences. Therefore ocular leishmaniasis
is considered potentially a blinding disorder and early diagnosis, by
maintaining a high level of suspicion by physicians who work in endemic
areas, and early treatment may prevent blinding complications. Three cases
have been reported that caused blindness [9]. Our case suggests that intralesional
injection of stibogluconate in addition to its systemic use, especially
when combined with allupurinol, could cause complete healing of eyelid
involvement in oriental sore and resolving of associated conjunctivitis
and episcleritis. Recurrence after the first two courses of treatment
in our patient was due to incomplete therapy. Failure in treatment might
have been due to both delay in diagnosis and incomplete therapy as well
as other factors such as resistant leishmania species or deficient host
immune responses. Early diagnosis and rigorous treatment is required in
order to prevent complications of this potentially blinding disorder.
Article accepted on 20/7/01
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