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Case Reports and Small Case Series
June 2000

Mucosal Leishmaniasis Presenting as Sinusitis and Optic Neuropathy

Arch Ophthalmol. 2000;118(6):852-854. doi:

Leishmaniasis, caused by the dismorphic protozoa Leishmania, is typically observed in patients as 1 of 3 clinical syndromes. Cutaneous leishmaniasis with chronic nonhealing ulcers that involve the face and extremities is the most common manifestation. Mucosal leishmaniasis invades the nose, oral cavity, and pharynx. Visceral leishmaniasis is characterized by fever, wasting, hepatosplenomegaly, and pancytopenia.1,2 To date, the only ocular problem has been eyelid involvement secondary to adjacent facial cutaneous infiltration.3,4 In view of the recent association between leishmaniasis and acquired immunodeficiency syndrome (AIDS),5 additional ophthalmological manifestations may be anticipated. We describe a unique observation of leishmaniasis infiltration of the orbital apex.

Report of a Case

A 45-year-old man was hospitalized for evaluation of a right-sided headache of 4 weeks' duration and 1 week of progressive graying of vision in his right eye . His medical history was remarkable for human immunodeficiency virus infection for approximately 14 years and recent diagnosis of AIDS due to the development of pulmonary Pneumocystis carinii pneumonia and multidrug-resistant tuberculosis (CD4 cell count<20/µL).

Findings from neuro-ophthalmologic examination revealed a visual acuity of 20/50-2 OD and 20/20 OS. He could not see the control or the test plates of the Ishihara color plates test with his right eye, but he saw all the plates with his left eye. On tangent perimetry at 1 m he could only count fingers in all quadrants with his right eye, and with the left eye the field was normal to 2 mm white object. In the right eye, there was 2 mm of exophthalmos without ptosis, swelling, conjunctival hyperemia, or limitation of ocular motility. The pupils were round and reactive to light; the right pupil was less reactive with a relative afferent pupillary defect. Results of slitlamp biomicroscopy and ophthalmoscopy were unremarkable.

Computed tomography demonstrated a sphenoid sinus lesion that extended into the right orbital apex through an area of bone erosion (Figure 1). Magnetic resonance imaging revealed diffuse opacification of the sphenoid sinus with areas of bright and decreased signals on T2-weighted images suggestive of fungal infection (Figure 2). Hematoxylin-eosin staining of the specimen obtained by endoscopic sphenoidectomy revealed intracellular organisms in the nasal and sphenoidal mucosa. Giemsa stain also demonstrated the intracellular organisms (Figure 3) while the Gomori methenamine-silver stain was negative for organisms. Gram stain did not show evidence of additional organisms. Fungal cultures of the biopsy specimen were also negative for organisms. A diagnosis of leishmaniasis was made and confirmed by the Centers for Disease Control and Prevention and the species identified as Leishmania braziliensis.

Figure 1.
Axial view of bone window computed tomographic scan with right sphenoid sinus opacity extending through bone defect to the right orbit. Arrow demonstrates the infiltration into the right cavernous sinus.

Axial view of bone window computed tomographic scan with right sphenoid sinus opacity extending through bone defect to the right orbit. Arrow demonstrates the infiltration into the right cavernous sinus.

Figure 2.
T2-weighted magnetic resonance imaging scan with a bright signal (arrow) of sinus mucosa and one area of decreased signal, extending to the right orbit.

T2-weighted magnetic resonance imaging scan with a bright signal (arrow) of sinus mucosa and one area of decreased signal, extending to the right orbit.

Figure 3.
Giemsa-stained tissue biopsy specimen demonstrating the intracellular Leishmania(arrows) (original magnification × 400).

Giemsa-stained tissue biopsy specimen demonstrating the intracellular Leishmania(arrows) (original magnification × 400).

The patient was treated with intravenous Pentostam (a European orphan drug; sodium stibagluconate) 1200 mg/d. Despite treatment, the vision in his right eye deteriorated to no light perception, and right third, fourth, and sixth cranial nerve palsies appeared. Additional computed tomography revealed progression of the process at the orbital apex. Simultaneously he developed a severe pneumonia caused by Aspergillus and 2 months following the diagnosis he was hospitalized for seizures and hemiparesis and died the next day. An autopsy was not permitted.

Comment

Leishmaniasis is caused by an obligatory intracellular parasite that is frequently transmitted through the bite of infected sand flies from an animal reservoir. Other modes of transmission including parenteral, congenital, and sexual have been reported. Mucosal leishmaniasis develops in less than 5% of patients, typically months to years following localized cutaneous leishmaniasis. Most cases of mucosal leishmaniasis are associated with L braziliensis, commonly referred to as American leishmaniasis. The process often starts in the nasal septum and results in perforation. The diagnosis is made by demonstration of parasites by Giemsa stain on biopsy specimens from affected tissue. Newer methods include antigen detection, polymerase chain reaction detection, and skin test. Mucosal leishmaniasis tends to be a chronic progressive disease that responds poorly to the treatment. Pentostam (20 mg/ kg) is used as first-line therapy for leishmaniasis and amphotericin B (0.5-1 mg/kg) as second-line treatment.1,2 In patients with AIDS, the compromised immune status contributes to the dissemination of the infection and atypical presentation.5

Patients with the human immunodeficiency virus commonly experience sinusitis, typically due to bacteria, but sinus infections and orbital involvement have been described with Aspergillus species, Pseudallescheria boydii, microsporidia, and Alternaria species. Non-Hodgkin lymphoma should also be considered in the differential diagnosis of sinus disease in a patient infected with human immunodeficiency virus.6,7 We could not find another case of sinusitis and optic neuropathy due to leishmaniasis. This case should alert ophthalmologists to the possibility of unusual infectious agents as the cause of orbital apex disorder in patients with AIDS.

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Article Information

Corresponding author: Mark J. Kupersmith, MD, Institute for Neurology and Neurosurgery, Beth Israel Medical Center, North Division, 170 East End Ave, New York, NY 10128 (e-mail: mkuper@bethisraelny.org).

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Meyer  RDGaultier  CRYamashita  JT  et al.  Fungal sinusitis in patient with AIDS: report of 4 cases and review of the literature. Medicine (Baltimore). 1994;7369- 78Article
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