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Editor,—American tegumentary leishmaniasis is an endemic disease occurring in Latin America, especially in Brazil.1 The agent is the protozoanLeishmania, transmitted by sand flies.Leishmania v braziliensis causes the mucocutaneous form of the disease, in which systemic dissemination to mucous membranes follows the primary ulcerative skin lesion.
We report a patient with nasal and conjunctival mucous leishmaniasis who developed an epidermoid carcinoma in the orbit.
A 58 year old man presented with a painless conjunctival mass of 2 months' duration. He reported nasal and mouth wounds and nasal flattening for 3 years and had a depressed scar in his right leg caused by an ulcer that developed 6 years before. He had been treated with N-methyl glucamine antimoniate 2 years before and underwent nasal reconstruction 1 year later. The patient had been a smoker and an alcoholic for 30 years and lived in an endemic region for tegumentar leishmaniasis.
Slit lamp examination revealed a lower left conjunctival nodule of 3×1 cm (Fig 1A) and ipsilateral dacryocystitis. The remainder of the ocular examination was normal. Leishmanin skin test and serology were positive. Biopsies of the conjunctival, mouth, and nasal lesions showed a chronic inflammatory process rich in plasmocytes and an immunohistochemical test was positive forLeishmania (Fig 1C). Dacryocystography disclosed left lacrimal duct stenosis and skin fistulisation, left maxillary sinus opacification, and nasal septum destruction. The patient was treated with intravenous amphotericin B (total dose 2500 mg), with reduction of the conjunctival lesion (Fig 1B). Immunohistochemistry in a new biopsy was negative.
The patient was lost to follow up for 2 years, when he showed up with left proptosis and destruction of the internal ocular commissure (Fig 1D). Computed tomography revealed a solid mass eroding the walls of the left maxillary sinus, zygomatic arch, and orbital floor (Fig2A). A biopsy of the maxillary tumour disclosed epidermoid carcinoma (Fig 2B). The patient underwent left maxillectomy and orbit exenteration. One year after surgery, the patient presented with partial dehiscence of the frontal flap, a local biopsy showing carcinoma recurrence. Face magnetic resonance did not show signs of tumour. The patient did not return to the hospital.
The upper airway is the most affected site in mucocutaneous leishmaniasis (MCL), and extensive destruction of nasal mucous membranes and cartilage, invasion of the face sinuses, oral cavity, larynx, and pharynx may occur. Diagnosis is confirmed by the demonstration of intracellular leishmania amastigotes in Giemsa stained slit skin smears, although the parasite may be difficult to find in chronic lesions. Immunohistochemistry for leishmania antigens is an important diagnostic tool. Histopathological analysis discloses features ranging from inflammatory infiltration of mononuclear cells and neutrophils to a granulomatous reaction.2 Pentavalent antimony is the drug of choice for the treatment of all types of leishmaniasis. In resistant cases, amphotericin B or pentamidine isothionate is indicated.
Cutaneous leishmaniasis of the eyelid is the most common ophthalmological finding in oriental and occidental tegumentary leishmaniasis,3 and conjunctival involvement may appear as an associated4 or isolated finding.5 Other ocular features in MCL include interstitial keratitis,6iridocyclitis,7 and chronic dacryocystitis.8
In the case reported here, the malignant tumour may have been present in the initial condition, but the diagnosis of MCL and the favourable response to treatment meant that a more extensive search for an additional diagnosis was not carried out. The development of a neoplasm at the site of a previous dermal scar is a well recognised phenomenon. Basal cell carcinomas have been reported to arise from a previous cutaneous leishmaniasis lesion.9 10 To our knowledge, this is the first case of development of a squamous cell carcinoma from a previous mucous leishmania lesion with major involvement of the eye.
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Capsule staining and mature cataracts: a comparison of indocyanine green and trypan blue dyes. D F Chang