1799 A Rare Cause of Dysphagia-Mucosal Leishmaniasis

Abstract

INTRODUCTION: Leishmaniasis is a parasitic disease spread by the bite of infected sand flies. Depending on the location of tissue or organ involvement, it may be described as cutaneous, mucosal or visceral. We present a case of mucosal leishmaniasis, exclusively involving the upper aerodigestive tract with primary symptom of dysphagia. This pattern of anatomical involvement is uncommon, and tissue biopsies may be non-specific, leading to delays in diagnosis and treatment. In our case immunohistochemical staining, molecular studies and clinical suspicion were required for the diagnosis. CASE DESCRIPTION/METHODS: A 42-year-old Hispanic man presented to the clinic with a 4-month history of progressive dysphagia. He denied sinonasal or respiratory symptoms. He had normal vital signs. Oral examination revealed a large painless exophytic mass replacing the soft and hard palates, extending to the hypopharynx (Figure 1). A CT scan of the neck with contrast showed a severe infiltrative process throughout the upper aerodigestive tract extending from the oral cavity to the subglottic airway (Figure 2). Repeat biopsies were non-diagnostic initially, showing pseudoepitheliomatous hyperplasia without evidence of dysplasia or neoplasia. Work up for endemic fungal, tuberculous and non-infectious granulomatous etiologies were negative. Further histopathologic review led to immunohistochemical staining of biopsies and assays which were positive for Leishmania panamensis. He was treated with oral miltefosine. At a 3-month follow up, oropharyngeal lesions had substantially regressed (Figure 3) and his dysphagia resolved. Sustained improvement was noted at a 1-year follow up. DISCUSSION: Mucosal leishmaniasis may be caused by different species, including Leishmania braziliensis and panamensis. It results from lymphangitic or hematogenous spread of leishmania parasites, usually from a pre-existing skin lesion. Symptoms of dysphagia or odynophagia may be the primary manifestation. Lesions are mostly exophytic and commonly involve the tongue, palate, oropharynx or larynx. Diagnosis requires demonstration of leishmania amastigotes or positive immunohistochemical staining in biopsy samples. Different treatment modalities may be used. Miltefosine is the latest drug treatment approved by the FDA for mucosal leishmaniasis. A high index of suspicion is required for the diagnosis of mucosal leishmaniasis as it potentially mimics other granulomatous, neoplastic and infectious etiologies of the upper aerodigestive tract.