Ocular Leishmaniasis Treated by Intralesional Amphotericin B.

Leishmaniasis is a group of diseases with varied clinical manifestations. Ocular involvement is an unusual presentation of leishmaniasis, and the eyelid is not a common site of cutaneous lesions, likely due to the mobility of the lids. Some case reports of conjunctival involvement are either a contiguous dissemination from lid margin or in the setting of disseminated leishmaniasis in an immunocompromised host. To our knowledge, isolated involvement of the bulbar conjunctiva has not been reported. We present the first case in the literature of a patient with an erythematous fibrovascular lesion in the interpalpebral zone that was clinically diagnosed as pterygium, but recurred at the site of surgical excision. After histopathologic diagnosis, the lesion was treated with intralesional injection of amphotericin B and improved completely within a few weeks. An accurate diagnosis of leishmaniasis in the eye may be challenging in many clinical settings. To our knowledge, an isolated pterygium.like lesion has not been reported in literature. In addition, intralesional injection of amphotericin B is a novel treatment method in this setting.

INTRODUCTION

Leishmaniasis is a group of protozoan diseases caused by several species of the genus Leishmania. Each species is endemic in a particular zoogeographical area and tends to cause a similar clinical presentation. However, variations of these general trends can occur. Human leishmaniasis is usually classified as cutaneous, mucocutaneous, and visceral. Mucocutaneous leishmaniasis is endemic in Central and South America and is usually caused by parasites of Leishmania braziliensis complex. After cutaneous involvement with these parasites, mucosal or mucocutaneous metastatic lesions may complicate the clinical picture.1 Metastatic mucosal involvement with other Leishmania species is otherwise uncommon, and direct involvement of mucosal surfaces with dermotropic species is also a rare possibility, as most mucosal surfaces are not exposed to a sand fly bite. The state of Khorasan Razavi in Northeastern Iran is an endemic focus for cutaneous leishmaniasis, and the prevalent species are Leishmania tropica (the majority) and Leishmania major. The involvement of the eye with these species is unusual and commonly limited to eyelid skin. Exceptional cases of conjunctival involvement are mentioned in literature, most of them either as a contiguous dissemination from lid margin or in the setting of a disseminated leishmaniasis in an immunocompromised host.2 We represent the first case in English, peer-reviewed literature of isolated bulbar conjunctival involvement with old world cutaneous leishmaniasis and treatment with intralesional injection of amphotericin B.

CASE REPORT

A 54-year-old healthy female attended an ophthalmology clinic with a few months history of a symptomatic lesion in the nasal bulbar conjunctiva within the interpalpebral zone in the right eye. On examination, there was an erythematous fibrovascular lesion almost 5 mm long. Initially, it was diagnosed as a pterygium. Due to a foreign body sensation and discomfort, surgical removal was performed. Histopathology of excised tissue showed granulomatous inflammation consisting of aggregates of epithelioid and giant cells through the stroma; some epithelioid cells were loaded with numerous Leishmania parasites. There was remarkable lymphohistiocytic infiltrate around granuloma foci [Figure 1a and b].

Figure 1

Histopathology of the excised conjunctival lesion (a) Low power, (b) High power

During follow-up over a few weeks, the lesion recurred at the surgical site. When the patient was asked about leishmaniasis lesions in herself or family members, she mentioned a small papule of leishmaniasis on her ankle a few months ago, which had been treated with intralesional injections of glucantime.

We elected to try intralesional injections of amphotericin B because, at the time of diagnosis, glucantime for systemic administration was unavailable, and the lesion was very small.

The amphotericin vial was prepared in the following manner: 10 ml of distilled water was used to dissolve 50 mg of amphotericin B (Phosome; Cipla Limited., Mumbai, India), and 0.3 ml of this solution was injected into the lesion. The administered dose was 1.5 mg per injection.3

Weekly injections of amphotericin B were performed by an Ophthalmologist Minnesota. The injections resulted in marked healing of the lesion within 6 weeks [Figure 2a-d]. There were minimal side effects that included a mild burning sensation and transient chemosis.

Figure 2

Clinical course of conjunctival lesion (a) Before treatment, (b) After 6 weeks of intralesional amphotericin injection, (c) 6 weeks after the last treatment session, (d) 6 months after treatment

DISCUSSION

Ocular leishmaniasis may be a significant diagnostic challenge and lead to delayed diagnosis or inappropriate treatments. The eyelid is the most common site of ocular involvement from case reports in literature.4 Periocular involvement with leishmaniasis is not common and comprises approximately 2–5% of the cases of facial leishmaniasis, likely due to movements of the eyelids which make fly bites very improbable. Papular, noduloulcerative type, plaque, and a nodular type of ocular leishmaniasis have been described. Some cases of dacryocystitis have been mentioned. O’Neill et al. reported a case of leishmaniasis of the eyelid in a patient with neurogenic ptosis and attributed it to immobility of the lid.5 Some cases have been reported of leishmaniasis lesions of the lid mimicking basal cell carcinoma and an infundibular cyst.6 In addition, some cases have been reported of leishmaniasis lesions of the lid margin and resembling chalazia.7 Lagophthalmos and blepharoconjunctivitis have also been reported in some lesions. The size of the lesions and their vicinity to the marginal free edge of the eyelid may be important in the occurrence of these symptoms.8 Cases of disseminated cutaneous leishmaniasis in immunocompromised hosts with multiple chalazia or hordeolum-like lesions of the conjunctiva and lid margin have also been reported. A similar clinical presentation has been described in postkala-azar dermal leishmaniasis (PKDL), in patients treated for visceral leishmaniasis. A report by el Hassan et al. reported conjunctivitis and blepharitis in 4 patients with PKDL, and Leishmania donovani was identified with polymerase chain reaction.3 In 2 other patients, they reported anterior uveitis in the course of PKDL.3 Gontijo et al.9 reported a case of cutaneous, visceral, and ophthalmic leishmaniasis in a kidney transplant patient. The case presented with skin lesions of cutaneous leishmaniasis, fever, and organomegaly; and developed conjunctival hyperemia, intense ocular pain, and low vision. L. (Viannia) braziliensis was isolated from the iliac crest, aqueous humor, and vitreous body.9 A 58-year-old male with American mucocutaneous leishmaniasis developed interstitial keratitis and was treated with systemic amphotericin B.10 Ocular involvement of our patient resembled pterygium which recurred rapidly after surgical removal. To our knowledge, no other similar case has been reported in English peer- reviewed literature.

Treatment of ocular leishmaniasis is more challenging. Systemic antimoniate (e.g. Glucantime) is the first-line modality in leishmaniasis treatment. Amphotericin has been shown to be effective in the treatment of leishmaniasis.1 We have previously reported successful treatment with systemic amphotericin B in an immunocompromised patient with disseminated cutaneous leishmaniasis and ocular involvement.11 Recently, we showed that intralesional amphotericin B is a good alternative for patients who are resistant to antimoniate therapy or allergic to antimoniates.12

In ophthalmology, amphotericin B has been used by systemic, topical (eyedrops and ointment), intracameral, intrastromal, and intravitreal routes in the treatment of fungal keratitis caused by Aspergillus spp. Fusarium spp., and Candida spp. Amphotericin B is the most common topical treatment for yeast and the second most common treatment for filamentous keratitis. Amphotericin B is available as a powder and is diluted in 5% dextrose as a solution for topical or subconjunctival administration. In the current case, we applied intralesional amphotericin B in concentrations similar to subconjunctival route.

To our knowledge, this is the first case of ocular leishmaniasis treated with intralesional (subconjunctival) amphotericin B, which led to reasonable efficacy and tolerable side effects.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.