Nodular Lymphangitis a Common Presentation of Cutaneous Leishmaniasis in Panama Capitan-Barrios, Z.; Nieto, J.; Nieto, M.; Sosa, N. Gorgas Memorial Institute for Health Studies, Rep. of Panama American tegumentary leishmaniasis (ATL) is one of the most prevalent parasitic zoonosis in Panama, and a major health problem with approximately 3,000 new cases per year. The infection is associated with a variety of clinical presentations, depending mainly on the species of the parasite and the host immune response. The typical skin manifestations are characterized by a single or few ulcerative lesions that are shallow and circular with well-defined borders and a bed of granulation tissue. Recently, there has been an increase in the number of reports of rare and atypical variants of ATL. We described the clinical, laboratorial and epidemiological features of four male patients with ATL (two with 16 years of age, one patient with 25 years of age and one patient with 26 years of age) with the sporotrichoid lymphocutaneous variety also known as nodular lymphangitis, characterized by the presence of multiple subcutaneous nodules along the lymphatic vessels draining a primary skin ulcer. These 4 cases were identified in a group of 25 consecutive patients enrolled in a therapeutic trial currently being conducted at Gorgas Memorial Institute (GMI). The average number of ulcerated skin lesions on these 4 patients was 3, ranging from 2 - 5 lesions. The average duration of their cutaneus condition was 1.4 months. ATL diagnosis was confirmed by traditional methods (smears, culture and Montenegro Test) and the identification of the parasite was done using molecular tests. Using PCR-RFLP analysis and direct sequencing, Leishmania Viannia panamensis was confirmed as the species responsible for these infections. Our results suggest that sporotrichoid lymphocutaneous leishmaniasis, or nodular lymphagitis is another relatively common clinical presentation of ATL in Panama, where L.V. panamensis is predominant. Case Number/age/gender Case 1. / 16 years / Male Case 2. / 25 years / Male Case 3. / 16 years / Male Case 4. / 26 years / Male C # of Ulcerated Time of Evolution of the Endemic Area Figure Lesions/Anatomic Location Lesions 5 / right forearm 3 months Capira/Province of Panama C 1 month 2 / right wrist Capira/Province of Panama D 2 / neck and left forearm 2 months Province of Darien A, E, F Cerro Azul/Prov. of Panama B, G, H, I and J 3 months 2 / neck and left elbow D L L Cutaneous leishmaniasis is a neglected disease with approximately 12 million people in- fected in 88 tropical and subtropical countries. In Panama, ATL is a major health problem, with an estimated 3,000 new cases per year (60–100 new cases per 100,000 habitants), with a probable 50% rate of under reporting. Leishmaniasis is associated with a variety of clinical manifestations, depending on the species of the parasite, the host immune response, and factors in the saliva of the sand fly vector. In Panama, four Leishmania species potentially pathogenic to humans have been described (L. panamensis, L. mexicana, L. amazonensis, and L. colombiensis). Among these, L. (Viannia) panamensis is the most widespread species and is responsible for the majority of human cases in the country (Fig A and B). A C. Case 1, nodular lymphangitis near to lesion in right forearm. D. Case 2, nodular lymphangitis near to lesion in right wrist . L = ulcerated lesion F E B L L E, F. Two different pictures of the nodular lymphangitis in the left forearm of the patient case 3 . L = ulcerated lesion G H A. Lesion of Leishmaniasis in left forearm. B. Lesion of Leishmaniasis in elbow. Sporotrichoid lymphocutaneous infection or Nodular lymphangitis (NL) describes a charac- teristic clinical presentation in which erythematous papules and nodules are located along the site of lymphatic drainage; it is a syndrome that is often misdiagnosed and improperly treated. The majority of the cases of NL in the United States are caused by Sporothrix schenckii, Nocardia brasiliensis, Mycobacterium marinum or Leishmania spp. The sporotrichoid disease begins at a site of distal inoculation and leads to the development of nodular lymphangitis. Sporotrichoid leishmaniasis is a sporadic form of cutaneous leishmaniasis. Clinically, it occurs as nontender, subcutaneous, slightly erythematous nodules, often associated with lymphangitis, usually on exposed areas of the skin. L I J We present 4 cases of documented ATL in Panama with NL (table 1). In all four cases the diagnosis was confirmed by traditional methods (smears, culture and Montenegro Test) and the identification of the parasite was done using molecular tests. Using PCR-RFLP analysis and direct sequencing, Leishmania Viannia panamensis was confirmed as the species responsible for these infections. These cases were part of a group of 25 consecutive patients enrolled in a ATL therapeutic trial (frecuency of 16% of NL among this patients). All the four cases had distal ulcerated lesions and subcutaneous nodules in the distribution of the lymphatic vessels. Only case 4 developed pain and discomfort in the area of the nodules. One of these subcutaneous nodules became an abscess and were aspirated with a syringe to rule out secondary bacterial infection (Fig I). Approximately 1.5mL of pus was sent for Gram stain, culture, PCR for Leishmania spp. and Leishmania spp. culture. The Gram stain, bacterial and Leishmania cultures were negative, but the PCR for Leishmania spp. was positive. NL is a relatively common presentation of ATL in Panama and it should be considered in the differential diagnosis of patients with sporotrichoid lesions, in whom this etiology diagnosis may not be considered at first. G, H, I and J. Nodular lymphangitis in the left arm of the patient. Red arrow indicates the aspirated nodule. L = ulcerated lesion This is to our knowledge the first report of NL in Panama. This clinical presentation has been reported elsewhere. In a report by Rodriguez EM et al, a higher incidence (43.3% 198/457 cases) of NL in ATL was found in Venezuela. -Cozzani E, et al. 2010. CED doi: 10.1111/j.1365-2230.2010.03855.x -Miranda A, et al. 2009. Am. J. Trop. Med. Hyg., 81(4):565–571 -Ellis HT, et al. 2001. American Family Phisician 63(2):326-332 -Rodriguez EM, et al. 2001. The 129th Annual Meeting of APHA -Smego RA, et al. 1999. Medicine. 78(1), 38-63 ZCB was supported by Gorgas Memorial Institute in Panama and The Office of The Surgeon General, Department of the US Army
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