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Ocular scleromalacia caused by leishmaniasis: a rare cause of scleral perforation
  1. P REINECKE,
  2. H E GABBERT
  1. Department of Pathology, University Hospital
  2. D-40225 Düsseldorf, Germany
  3. Department of Ophthalmology, Evangelisches Hospital D-45466 Mülheim/Ruhr, Germany
  1. W STRUNK,
  2. C C LÖSCHE
  1. Department of Pathology, University Hospital
  2. D-40225 Düsseldorf, Germany
  3. Department of Ophthalmology, Evangelisches Hospital D-45466 Mülheim/Ruhr, Germany
  1. Petra Reinecke, MD, Institute of Pathology, Heinrich-Heine-University, Moorenstrasse 5, D-40225 Düsseldorf, Germany

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Editor,—The clinical manifestations of leishmaniasis depend on complex interactions between the virulence, characteristics of the infecting Leishmaniaspecies, and the immune response of its host.1-3Leishmaniasis sometimes involves the eye as a result of contagious spread from the eyelid and conjunctiva, by the haematogenous route, or by inoculation of the conjunctiva by the patient's own fingers, usually caused by L donovani.4-7

We report a case of an Afghan boy suffering from general mucocutaneous leishmaniasis caused by L tropica and a bilateral intraocular manifestation of this disease.

CASE REPORT

An 11 year old Afghan boy with known general mucocutaneous leishmaniasis caused by L tropica stayed in the “Friedensdorf International”, Oberhausen, Germany, for treatment of his disease. After he had undergone a systemic eradication therapy in the summer of 1995 (Institute of Tropical Medicine, University of Tuebingen, J Knobloch and the Paul Lechler Hospital for Tropical Diseases, Tuebingen, Germany), the boy was first seen in the Department of Ophthalmology, Mülheim/Ruhr, Germany, in January 1996 with binocular uveitis and secondary cataract. The right eye showed only slight inflammation, but the left eye severe inflammation of the anterior chamber. The cataract was more advanced in the right eye, visual acuity reduced to light perception in the right eye, to 0.2 in the left. Local anti-inflammatory therapy was started and cataract surgery was intended. The patient was seen again in spring 1996. Both eyes were painful, visual acuity in the left eye had also decreased to light perception, inflammation had severely increased with granulomata and vascularisation on the iris and narrowing of the anterior chamber in both eyes. After eradication therapy reinfection was thought to be very unlikely at this time but the patient deteriorated rapidly. Severe scleromalacia appeared in both eyes near the superior limbus followed by scleral perforation of the right eye. Severe pain, loss of vision, the extent of perforation, and inflammation made the enucleation of the right eye inevitable (Fig 1A).

Figure 1

(A) Clinical examination of the right eye. Severe scleromalacia with focal perforation. (B) Macroscopical examination. Perforated sclera, lens, parts of the iris, and vitreous body luxated into the bulge. (C) Light microscopic examination. Leishmaniae (arrow) within histiocytes by Giemsa staining. (D) Transmission electron microscopic examination The protozoon consists of a nucleus (n), prominent mitochondria (m), and the flagellum (f).

Macroscopic examination showed the perforated sclera, with prolapse of the lens, parts of the iris, and the vitreous body (Fig 1B). The histopathological examination showed many histiocytes filled with encapsulated leishmania 2–4 μmol in length (Fig 1C). The diagnosis was confirmed by transmission electron microscopy showing frequent amastigote organisms within the cytoplasm of histiocytes, with a nucleus, a kinetoplast, and an intracellular flagellum (Fig 1D). After histological examination intensive anti-infectious therapy was started again. Despite all efforts, scleral perforation also occurred in almost the entire inferior part of the sclera near the limbus of the left eye, so enucleation of the left bulbus had to be performed. Intraoperatively, both orbits were without pathological findings and the postoperative recovery was without complications.

COMMENT

The estimated worldwide overall prevalence of leishmaniasis is 12 million with the population at risk approaching 350 million.1 Visceral leishmaniasis is typically caused byL donovani.3 Leishmaniasis of the eye is very rare and the few reported cases were either due toL donovani45 or theLeishmania subtype was not characterised.67Leishmania tropica was thought to cause cutaneous infection exclusively,1 but in a small group of American military personnel who served in Operation Desert Storm,L tropica was isolated from bone marrow specimens taken after the soldiers had developed chronic low fever, fatigue, and in some instances diarrhoea.8 This kind of leishmaniasis was named “viscerotropic leishmaniasis” (VTL) because the clinical symptoms differed in some points from the typical visceral leishmaniasis. In this boy's case the channel of transmission could not be determined. However, as no pathological findings of the eyelids could be observed the very rare haematogenous route seems to be the most likely mode of infection, possibly caused by the previously diagnosed L tropica being the cause of the generalised mucocutaneous leishmaniasis. Identifying the species ofLeishmania is especially important for the evaluation of treatment and their different sensitivities to anti-infectious drugs.1

References

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