Leishmaniasis is a protozoal infection with an increased risk of transmission to those serving in the U.S. Military due to theaters of operation in endemic regions. There has, in recent decades, been robust experience with old-world leishmaniasis in the Military Health System (MHS); however, new-world leishmaniasis, which may result in mucosal leishmaniasis, has been less studied. A total of 88 patients from 2012 to 2022 with diagnosis codes for "mucocutaneous leishmaniasis" or "leishmaniasis, unspecified" were identified in the Military Data Repository and reviewed. Within this cohort, upon medical records review, there were 2 cases of leishmaniasis that met inclusion criteria. Case 1 was a 28-year-old active duty male with recent travel to Belize who presented with a mucosal lip lesion that was biopsied and had inconclusive species confirmation but was thought to be either L. braziliensis or L. mexicana. The second case involved a 30-year-old active duty male with a history of travel to French Guiana who had a cutaneous lesion on his left hand that was identified as L. guyanensis, a causative species for mucosal leishmaniasis. Neither had evidence of any further mucosal involvement on otolaryngologic evaluation, and both subsequently received systemic therapy with a good clinical response. Although only 2 cases were identified over an 11-year period, this disease remains an important medical consideration when conducting military operations within Central and South America, as both cases had recent military-specific travel to areas endemic for leishmaniasis.
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