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Answer to Photo Quiz: Anal Leishmaniasis (See page 1675 in this issue [doi:10.1128/JCM.01529-14] for photo quiz case presentation.) Antonio Lalueza,a Félix Cambra,b María Urbanowicz,c Rafael San Juan,a Guadalupe López-Alonso,c José María Aguadoa Infectious Diseases Unit, University Hospital 12 de Octubre, Madrid, Spaina; Department of Surgery, University Hospital 12 de Octubre, Madrid, Spainb; Department of Pathology, University Hospital 12 de Octubre, Madrid, Spainc
G
iemsa-stained touch preparations of the anal biopsy material showed intracellular Leishmania amastigotes. The patient’s serologic analysis result for Leishmania was positive. Colonic histology showed no Leishmania, and a bone marrow biopsy showed no significant findings. Results for both culture and direct microscopic examination of bone marrow aspirate were negative for Leishmania. The patient was treated with 3 mg/kg of body weight of liposomal amphotericin B on days 1 to 5 daily and then on days 14 and 21 (with a total dose of 21 mg/kg). The patient had a partial clinical response to treatment, with disappearance of local symptoms but with hardly any reduction of the size of the lesion. Therefore, a surgical excision of the lesion was performed 3 months later. The staining and culture of the subsequent tissue sample showed no Leishmania. Leishmaniasis is a zoonotic disease, endemic in some Mediterranean countries, such as Spain (1). Leishmania is a facultative intracellular protozoan. The parasite infects the macrophages of its mammalian hosts, and hosts who are immunocompetent recover after development of effective cell-mediated immunity, which allows the activation of macrophages to a microbicidal state. A decrease in CD4⫹ cell count and reduced production of interferon, such as in HIV-positive patients, may facilitate the spread of Leishmania protozoa and diffuse visceral parasitization, thus resulting in unusual locations of these organisms (1). Atypical locations, such as with visceral leishmaniasis (VL) confined to the gastrointestinal tract alone, can occur, especially in HIV-positive patients (2); however, this condition is highly unusual in immunocompetent individuals (3). In patients with gastrointestinal leishmaniasis, the duodenal mucosa has been described as the region of the digestive tract most frequently affected (90% of cases) (4). Absence of hepatosplenomegaly is exceptional in VL (5, 6). Although rare, there have been reported cases of intestinal leishmaniasis without pancytopenia (7). However, there have been only a few reported cases of leishmaniasis with anal involvement, all of them in HIV-positive patients except for one case (8–11). We believe that a relative cellular immunosuppression unrelated to HIV in our case produced this atypical form of leishmaniasis with anal involvement without bone marrow infiltration or hepatosplenomegaly and without other sites of the gastrointestinal tract affected. In atypical cases, such as this one, distinguishing between the cutaneous and visceral forms of the disease can be difficult. However, systemic manifestations such as weight loss, mucocutaneous involvement in places not exposed to sandfly bite, and a positive serologic analysis result for Leishmania, as well as hypergammaglobulinemia, high erythrocyte sedimentation rate, circulating immune complexes like anti-smooth muscle antibody (12), and positive
July 2016 Volume 54 Number 7
rheumatoid factor (13), strongly suggest that this case fits with a VL form. In conclusion, in areas of endemicity, leishmaniasis should be considered possible in elderly patients with decreased CD4⫹ cell counts and an anal mass without evidence of tumor cells in the biopsy specimen. ACKNOWLEDGMENTS Sincere thanks to Alejandra Morales-Cartagena for the review of the manuscript. We declare no financial support. We declare no conflicts of interest.
REFERENCES 1. Herwaldt BL. 1999. Leishmaniasis. Lancet 354:1191–1199. http://dx.doi .org/10.1016/S0140-6736(98)10178-2. 2. Alvar J, Aparicio P, Aseffa A, Den Boer M, Canavate C, Dedet JP, Gradoni L, Ter Horst R, Lopez-Velez R, Moreno J. 2008. The relationship between leishmaniasis and AIDS: the second 10 years. Clin Microbiol Rev 21:334 –359. http://dx.doi.org/10.1128/CMR.00061-07. 3. Alvarez-Nebreda ML, Alvarez-Fernandez E, Rada S, Branas F, Maranon E, Vidan MT, Serra-Rexach JA. 2005. Unusual duodenal presentation of leishmaniasis. J Clin Pathol 58:1321–1322. http://dx.doi.org/10.1136/jcp .2005.027029. 4. Laguna F, Garcia-Samaniego J, Soriano V, Valencia E, Redondo C, Alonso MJ, Gonzalez-Lahoz JM. 1994. Gastrointestinal leishmaniasis in human immunodeficiency virus-infected patients: report of five cases and review. Clin Infect Dis 19:48 –53. http://dx.doi.org/10.1093 /clinids/19.1.48. 5. Hicks L, Kant P, Tay PH, Vincini V, Schuster H, Rotimi O, Maughan N, Jordan C, Moss S, Everett S, Hamlin PJ. 2009. Visceral Leishmaniasis presenting with intestinal failure: a case report and literature review. Eur J Gastroenterol Hepatol 21:117–122. http://dx.doi.org/10 .1097/MEG.0b013e32830e6fdb. 6. Parija SC, Karki P, Koirala S. 2000. Cases of kala-azar without any hepatosplenomegaly. Trop Doct 30:187–188. 7. Datry A, Similowski T, Jais P, Rosenheim M, Katlama C, Maheu E, Kazaz S, Fassin D, Danis M, Gentilini M. 1990. AIDS-associated leishmaniasis: an unusual gastro-duodenal presentation. Trans R Soc Trop Med Hyg 84:239 –240. http://dx.doi.org/10.1016/0035-9203(90)90269-K. 8. Armengot-Carbo M, Carmena-Ramon R, Rodrigo-Nicolas B, FerrandoMarco J. 2012. Unsuspected visceral leishmaniasis infiltrating a squamous cell carcinoma. Actas Dermosifiliogr 103:321–323. http://dx.doi.org/10 .1016/j.ad.2011.04.016. 9. Hernandez DE, Oliver M, Martinez C, Planas G. 1995. Visceral leishmaniasis with cutaneous and rectal dissemination due to Leishmania braziliensis in acquired immunodeficiency syndrome (AIDS). Int J Dermatol 34:114 –115. http://dx.doi.org/10.1111/j.1365-4362.1995.tb03592.x.
Citation Lalueza A, Cambra F, Urbanowicz M, San Juan R, López-Alonso G, Aguado JM. 2016. Answer to photo quiz: anal leishmaniasis. J Clin Microbiol 54:1935–1936. doi:10.1128/JCM.01530-14. Editor: P. Bourbeau Address correspondence to Antonio Lalueza, [email protected]. Copyright © 2016, American Society for Microbiology. All Rights Reserved.
Journal of Clinical Microbiology
jcm.asm.org
1935
Photo Quiz
10. Idoate MA, Vazquez JJ, Civeira P. 1993. Rectal biopsy as a diagnostic procedure of chronic visceral leishmaniasis. Histopathology 22:589 –590. http://dx.doi.org/10.1111/j.1365-2559.1993.tb00182.x. 11. Rosenthal PJ, Chaisson RE, Hadley WK, Leech JH. 1988. Rectal leishmaniasis in a patient with acquired immunodeficiency syndrome. Am J Med 84:307–309. http://dx.doi.org/10.1016/0002-9343(88) 90430-5.
1936
jcm.asm.org
12. Argov S, Jaffe CL, Krupp M, Slor H, Shoenfeld Y. 1989. Autoantibody production by patients infected with Leishmania. Clin Exp Immunol 76: 190 –197. 13. Atta AM, Carvalho EM, Jeronimo SM, Sousa Atta ML. 2007. Serum markers of rheumatoid arthritis in visceral leishmaniasis: rheumatoid factor and anti-cyclic citrullinated peptide antibody. J Autoimmun 28:55–58. http://dx.doi.org/10.1016/j.jaut.2006.12.001.
Journal of Clinical Microbiology
July 2016 Volume 54 Number 7
Answer to Photo Quiz: Anal Leishmaniasis (See page 1675 in this issue [doi:10.1128/JCM.01529-14] for photo quiz case presentation.) Antonio Lalueza,a Félix Cambra,b María Urbanowicz,c Rafael San Juan,a Guadalupe López-Alonso,c José María Aguadoa Infectious Diseases Unit, University Hospital 12 de Octubre, Madrid, Spaina; Department of Surgery, University Hospital 12 de Octubre, Madrid, Spainb; Department of Pathology, University Hospital 12 de Octubre, Madrid, Spainc
G
iemsa-stained touch preparations of the anal biopsy material showed intracellular Leishmania amastigotes. The patient’s serologic analysis result for Leishmania was positive. Colonic histology showed no Leishmania, and a bone marrow biopsy showed no significant findings. Results for both culture and direct microscopic examination of bone marrow aspirate were negative for Leishmania. The patient was treated with 3 mg/kg of body weight of liposomal amphotericin B on days 1 to 5 daily and then on days 14 and 21 (with a total dose of 21 mg/kg). The patient had a partial clinical response to treatment, with disappearance of local symptoms but with hardly any reduction of the size of the lesion. Therefore, a surgical excision of the lesion was performed 3 months later. The staining and culture of the subsequent tissue sample showed no Leishmania. Leishmaniasis is a zoonotic disease, endemic in some Mediterranean countries, such as Spain (1). Leishmania is a facultative intracellular protozoan. The parasite infects the macrophages of its mammalian hosts, and hosts who are immunocompetent recover after development of effective cell-mediated immunity, which allows the activation of macrophages to a microbicidal state. A decrease in CD4⫹ cell count and reduced production of interferon, such as in HIV-positive patients, may facilitate the spread of Leishmania protozoa and diffuse visceral parasitization, thus resulting in unusual locations of these organisms (1). Atypical locations, such as with visceral leishmaniasis (VL) confined to the gastrointestinal tract alone, can occur, especially in HIV-positive patients (2); however, this condition is highly unusual in immunocompetent individuals (3). In patients with gastrointestinal leishmaniasis, the duodenal mucosa has been described as the region of the digestive tract most frequently affected (90% of cases) (4). Absence of hepatosplenomegaly is exceptional in VL (5, 6). Although rare, there have been reported cases of intestinal leishmaniasis without pancytopenia (7). However, there have been only a few reported cases of leishmaniasis with anal involvement, all of them in HIV-positive patients except for one case (8–11). We believe that a relative cellular immunosuppression unrelated to HIV in our case produced this atypical form of leishmaniasis with anal involvement without bone marrow infiltration or hepatosplenomegaly and without other sites of the gastrointestinal tract affected. In atypical cases, such as this one, distinguishing between the cutaneous and visceral forms of the disease can be difficult. However, systemic manifestations such as weight loss, mucocutaneous involvement in places not exposed to sandfly bite, and a positive serologic analysis result for Leishmania, as well as hypergammaglobulinemia, high erythrocyte sedimentation rate, circulating immune complexes like anti-smooth muscle antibody (12), and positive
July 2016 Volume 54 Number 7
rheumatoid factor (13), strongly suggest that this case fits with a VL form. In conclusion, in areas of endemicity, leishmaniasis should be considered possible in elderly patients with decreased CD4⫹ cell counts and an anal mass without evidence of tumor cells in the biopsy specimen. ACKNOWLEDGMENTS Sincere thanks to Alejandra Morales-Cartagena for the review of the manuscript. We declare no financial support. We declare no conflicts of interest.
REFERENCES 1. Herwaldt BL. 1999. Leishmaniasis. Lancet 354:1191–1199. http://dx.doi .org/10.1016/S0140-6736(98)10178-2. 2. Alvar J, Aparicio P, Aseffa A, Den Boer M, Canavate C, Dedet JP, Gradoni L, Ter Horst R, Lopez-Velez R, Moreno J. 2008. The relationship between leishmaniasis and AIDS: the second 10 years. Clin Microbiol Rev 21:334 –359. http://dx.doi.org/10.1128/CMR.00061-07. 3. Alvarez-Nebreda ML, Alvarez-Fernandez E, Rada S, Branas F, Maranon E, Vidan MT, Serra-Rexach JA. 2005. Unusual duodenal presentation of leishmaniasis. J Clin Pathol 58:1321–1322. http://dx.doi.org/10.1136/jcp .2005.027029. 4. Laguna F, Garcia-Samaniego J, Soriano V, Valencia E, Redondo C, Alonso MJ, Gonzalez-Lahoz JM. 1994. Gastrointestinal leishmaniasis in human immunodeficiency virus-infected patients: report of five cases and review. Clin Infect Dis 19:48 –53. http://dx.doi.org/10.1093 /clinids/19.1.48. 5. Hicks L, Kant P, Tay PH, Vincini V, Schuster H, Rotimi O, Maughan N, Jordan C, Moss S, Everett S, Hamlin PJ. 2009. Visceral Leishmaniasis presenting with intestinal failure: a case report and literature review. Eur J Gastroenterol Hepatol 21:117–122. http://dx.doi.org/10 .1097/MEG.0b013e32830e6fdb. 6. Parija SC, Karki P, Koirala S. 2000. Cases of kala-azar without any hepatosplenomegaly. Trop Doct 30:187–188. 7. Datry A, Similowski T, Jais P, Rosenheim M, Katlama C, Maheu E, Kazaz S, Fassin D, Danis M, Gentilini M. 1990. AIDS-associated leishmaniasis: an unusual gastro-duodenal presentation. Trans R Soc Trop Med Hyg 84:239 –240. http://dx.doi.org/10.1016/0035-9203(90)90269-K. 8. Armengot-Carbo M, Carmena-Ramon R, Rodrigo-Nicolas B, FerrandoMarco J. 2012. Unsuspected visceral leishmaniasis infiltrating a squamous cell carcinoma. Actas Dermosifiliogr 103:321–323. http://dx.doi.org/10 .1016/j.ad.2011.04.016. 9. Hernandez DE, Oliver M, Martinez C, Planas G. 1995. Visceral leishmaniasis with cutaneous and rectal dissemination due to Leishmania braziliensis in acquired immunodeficiency syndrome (AIDS). Int J Dermatol 34:114 –115. http://dx.doi.org/10.1111/j.1365-4362.1995.tb03592.x.
Citation Lalueza A, Cambra F, Urbanowicz M, San Juan R, López-Alonso G, Aguado JM. 2016. Answer to photo quiz: anal leishmaniasis. J Clin Microbiol 54:1935–1936. doi:10.1128/JCM.01530-14. Editor: P. Bourbeau Address correspondence to Antonio Lalueza, [email protected]. Copyright © 2016, American Society for Microbiology. All Rights Reserved.
Journal of Clinical Microbiology
jcm.asm.org
1935
Photo Quiz
10. Idoate MA, Vazquez JJ, Civeira P. 1993. Rectal biopsy as a diagnostic procedure of chronic visceral leishmaniasis. Histopathology 22:589 –590. http://dx.doi.org/10.1111/j.1365-2559.1993.tb00182.x. 11. Rosenthal PJ, Chaisson RE, Hadley WK, Leech JH. 1988. Rectal leishmaniasis in a patient with acquired immunodeficiency syndrome. Am J Med 84:307–309. http://dx.doi.org/10.1016/0002-9343(88) 90430-5.
1936
jcm.asm.org
12. Argov S, Jaffe CL, Krupp M, Slor H, Shoenfeld Y. 1989. Autoantibody production by patients infected with Leishmania. Clin Exp Immunol 76: 190 –197. 13. Atta AM, Carvalho EM, Jeronimo SM, Sousa Atta ML. 2007. Serum markers of rheumatoid arthritis in visceral leishmaniasis: rheumatoid factor and anti-cyclic citrullinated peptide antibody. J Autoimmun 28:55–58. http://dx.doi.org/10.1016/j.jaut.2006.12.001.
Journal of Clinical Microbiology
July 2016 Volume 54 Number 7
