Dermatology Online Journal

Ocular leishmaniasis: A case report
Sadeghian G¹, Nilfroushzadeh MA², Moradi SH³, Hanjani SH⁴
Dermatology Online Journal 11 (2): 19

1. Assistant Professor of Dermatology, Skin Disease and Leishmaniasis Research Center. Isfahan University of Medical Sciences and Health Services, Isfahan, Iran. Sadeghian@mui.ac.ir 2. Assistant Professor of Dermatology Tehran university, Director of Skin Disease and Leishmaniasis Research Center, Isfahan, Iran. 3. Assistant Professor of Infectious and Tropical Diseases, Skin Disease and Leishmaniasis Research Center. Isfahan University of Medical Sciences and Health Services, Isfahan, Iran. 4. Ophtalmologist, Isabne Maryam Hospital, Isfahan University of Medical Sciences and Health Services, Isfahan, Iran.

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Abstract

Cutaneous leishmaniasis (CL) is a protozoal disease which is endemic in Iran usually caused by Leishmaniasis major and Leishmaniasis tropica and transmitted by the bite of a sandfly. In Isfahan province CL is highly prevalent and we observe some unusual clinical features of disease. The eyelid is rarely involved possibly because the movement of the lids prevents the fly vector from biting the skin in this region. We report a case of ocular leishmaniasis with eyelid and conjunctival involvement that had simulated chalazion and was complicated with trichiasis. The patient was diagnosed by direct smear, culture, and PCR from the lesions. He was treated with systemic sodium stibogluconate (20 mg/kg/day) for 20 days and subsequently surgery for trichiasis. The patient was clinically cured with this treatment, however the disease had left complications, including palpebral and conjanctival scaring, corneal opacity, and eyelash loss.

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The protozoon Leishmania, which is transmitted by the bite of a sandfly, can cause three distinct clinical entities: cutaneous leishmaniasis associated with Leishmania tropica in the old world and with subgenera Leishmania and Viannia in the new world; kala azar associated with Leishmania donovani and L.infantum; and mucocutaneous leishmaniasis associated with Leishmania braziliensis. In cutaneous leishmaniasis, the eyelid is involved in 0.4 percent of cases [1]. Clinical diagnosis of ocular leishmaniasis is very difficult and this disease may simulate other more common lesions such as chalazion, dacriocystitis, and tumors [2, 3, 4]. Conjunctiva are rarely affected [5]. We report a case of ocular leishmaniasis with eyelid and conjunctival involvement, associated with trichiasis, and which responded well to treatment with intramuscular sodium stibocoluconate preventing from further complications.

Clinical synopsis

A 13-year-old Afghanian boy referred to Skin Disease & Leishmaniasis Research Center with two red nodules at lower and upper eyelid margins. He had a history of cutaneous leishmaniasis on the nose 2 years prior, which had been treated with intralesional stiboglocunate weekly for 8 weeks. After the first 6 months he had been evaluated by an ophthalmologist for redness and induration on the right eyelids. The case was diagnosed as hordeolum and was treated with antibiotic eye drops and ointments, but no improvement was noticed. When we evaluated him we observed two hordeolum-like nodules, 5 mm X 5 mm and 10 mm X5 mm involving the upper and lower lids respectively. Also present was conjunctivitis and trichiasis. Two lymph nodes were palpable, one above the right eyebrow and other under lower lid. The patient was unable open the right eye (Fig. 1).

Figure 1	Figure 2
Fig 1: Clinical feature of the patient in the first visit.
Fig 2: Direct Smear was positive for leishman bodies (Gimsa stain)

Figure 3	Figure 4
Fig 3: Leishmania organism within macrophages (Haematoxylin & Eosin: magnification * 400)
Fig 4: Schisogram of amplified DNA related to isolated strain and standards by PCR method: 1-Unknown strain; 2-L.tropica (MHOM/SU/58/Strain/OD); 3- 1Kb Marker; 4- L.major (MRHO/IR/64/Nadim-1)

Figure 5
Clinical feature of the patient after a full course of treatment and 6-months followup

On the skin of his nose there was a red irregular scar. Routine physical examination and laboratory findings were within normal limits. Slit lamp examination revealed corneal abrasion attributed to trichiasis, but corneal stoma and sclera were normal. A direct smear of the lid lesions and bulbar conjunctiva revealed Leishman bodies (Fig. 2). Pathology examination showed leishmania organisms within and next to macrophages (Fig. 3). After isolation and culture of etiologic agent, PCR showed the isolated strain is related to a schisodeme of L. major. PCR was performed accompanying standard strains and 1 Kb base DNA standard marker (Fig. 4). The patient was diagnosed as suffering from ocular leishmaniasis and was treated with systemic sodium stiboglocunate for 20 days (20 mg/kg/day). Subsequent surgery for trichiasis was done by the ophtalmologist. After 2 months we observed a major improvement of lid erythema and induration, and the sporotrichoid nodules were resolving. A 6-month followup revealed clinical cure, but there wer residual complications including palpebral and conjunctival scaring, and eyelash loss of the right eye (Fig. 4). Ultimately visual acuity was 20/30 in the affected eye (because of opacity from exposure keratitis) and 20/20 in the left eye.

Discussion

Cutaneous leishmaniasis is a protozoal disease that is endemic in Iran; it is usually caused by L. major or L. tropica. Transmission of the disease occurs by the bite of a sandfly infected with leishmania parasites [6]. The disease in its various forms affects at least 12-million persons worldwide, with 400,000 new cases per year [7].

The eyelid is rarely involved in the cutaneous form of leishmaniasis, possibly the result of movement of the lids preventing the fly vector from biting the skin in this region [1]. It is very hard to establish a correct diagnosis of ocular 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, 3]. From 2 to 5 percent of the facial lesions are localized to the eyelids, most often on the lateral canthus. The pathologic pattern is comparable to what can be observed on the rest of the skin, but the fragility of eyelid results in a special risk of local spread [8].

In recent years a case of ocular leishamaniasis has been reported from Iran with clinical feature of chalasion-like lesion in lower lid, associated with conjunctivitis and nodular episcleritis. In that case recurrence had been noticed after treatment with both intramuscular and interalesional stibogluconate; ultimately the patient was cured with a combination of systemic sodium stibogluconate and alloporinol, and the lesions eventually healed with only small scar on the eye lid [5]. In addition, three cases of ocular leishmaniasis have been reported that have caused blindness. Two were caused by the direct extension of conjunctival leishmaniasis and one was caused by the systemic dissemination of kala-azar. Histologic sections of the enucleated eye showed multiple noncaseating granolomas with leishman bodies in the bulbar conjunctiva, iris, ciliary body, and retina [9].

Although cutaneous leishmaniasis is a self-limiting condition, if eyelid lesions remain untreated, the contiguous spread from the skin of the eyelid will extend to involve the conjunctiva, sclera, and even cornea, with development of interstitial keratitis [5]. Long-term complications of ocular leishmaniasis includes lid deformity with all its consequences. Therefore, ocular leishmaniasis is considered potentially a blinding disorder; early diagnosis by maintaining a high level of suspicion by physicians who work in endemic areas, and early treatment may prevent blinding complications [5]. In this case, the question persists whether the lid lesions were caused by the bite or by the inoculation of the lids by the patient's own fingers from the skin lesion of the nose [10], or by lymphatic dissemination considering the sporotrichoid nodules. The Koebner phenomenon (emergence of a new lesion after a mechanical trauma) must also be considered. Also regarding to the conjunctival involvement, the contiguous spread from skin of the eye lid had involved the conjunctiva over long time.

Other complications in this case were trichiasis, corneal opacity attributed to exposure keratitis, and eyelash loss following lid involvement. Our patient responded to systemic sodium stiboglucante and all the lesions resolved, however he was left with residual scars.

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