Leishmania Tropica–Induced Cutaneous and Presumptive Concomitant Viscerotropic Leishmaniasis with Prolonged Incubation

OBSERVATION Leishmania tropica–Induced Cutaneous and Presumptive Concomitant Viscerotropic Leishmaniasis With Prolonged Incubation Francesca Weiss, BS; Nicholas Vogenthaler, MD, MPH; Carlos Franco-Paredes, MD; Sareeta R. S. Parker, MD Background: Leishmaniasis includes a spectrum of dis- studies were highly suggestive of concomitant visceral eases caused by protozoan parasites belonging to the ge- involvement. The patient was treated with a 28-day course nus Leishmania. The disease is traditionally classified into of intravenous pentavalent antimonial compound so- visceral, cutaneous, or mucocutaneous leishmaniasis, de- dium stibogluconate with complete resolution of her sys- pending on clinical characteristics as well as the species temic signs and symptoms and improvement of her pre- involved. Leishmania tropica is one of the causative agents tibial ulcerations. of cutaneous leishmaniasis, with a typical incubation period of weeks to months. Conclusions: This is an exceptional case in that our patient presented with disease after an incubation period Observation: We describe a 17-year-old Afghani girl of years rather than the more typical weeks to months. who had lived in the United States for 4 years and who In addition, this patient had confirmed cutaneous in- presented with a 6-month history of pretibial ulcer- volvement, as well as strong evidence of viscerotropic dis- ations, 9.1-kg weight loss, abdominal pain, spleno- ease caused by L tropica, a species that characteristically megaly, and extreme fatigue. Histopathologic examina- displays dermotropism, not viscerotropism. tion and culture with isoenzyme electrophoresis speciation of her skin lesions confirmed the presence of L tropica. In addition, results of serum laboratory and serological Arch Dermatol. 2009;145(9):1023-1026 EISHMANIASIS INCLUDES A graphically into New World and Old World spectrum of disease caused disease. Old World disease is caused by by a group of protozoan Leishmania major, Leishmania tropica, Leish- parasites belonging to the ge- mania aethiopica, and, rarely, by Leish- nus Leishmania. Approxi- mania infantum, which are endemic in the Lmately 12 million people worldwide Middle East, India, and Africa. New World have some form of this disease, and more disease is caused by Leishmania brazilien- than 350 million people live in at-risk sis, Leishmania mexicana, or Leishmania areas.1 Indeed, Leishmania species infec- panamensis/Leishmania guyanesis com- tions, which are primarily transmitted by plexes and is endemic in some areas of the bite of infected sand flies, produce a Latin America.1 Clinical manifestations substantial burden of disease in more of CL vary from crusted verrucoid pap- than 88 countries worldwide.1 Leish- ules and plaques to disfiguring ulcer- maniasis presents with 3 major clinical ations. Lesions are typically located on ex- syndromes: visceral (VL), cutaneous posed body surfaces such as the lower legs (CL), and mucocutaneous leishmaniasis and arms. Mucocutaneous leishmaniasis is (ML).1 These clinical manifestations de- an immunopathogenic variant and is con- pend on complex host-parasite interac- sidered an oligoparasitic syndrome caused tions leading to primary replication of mainly by persistent L braziliensis disease. Author Affiliations: the parasite within macrophages in the Visceral leishmaniasis is predomi- Departments of Dermatology reticuloendothelial system in the case of nantly caused by Leishmania donovani or (Ms Weiss and Dr Parker) VL, in the dermis in CL, or in the naso- L infantum/Leishmania chagasi, and most and Infectious Disease 1 (Drs Vogenthaler and pharynx or oropharynx in ML. cases are concentrated in India (Bihar Franco-Paredes), Emory There are over 21 Leishmania species, State), Bangladesh, Sudan, and Brazil. University School of Medicine with each species having the potential to Clinical manifestations of VL include fe- and Grady Memorial Hospital cause more than 1 clinical syndrome.2,3 Cu- ver, weakness, weight loss, hepatospleno- Atlanta, Georgia. taneous leishmaniasis may be divided geo- megaly, pancytopenia, and hypergamma- (REPRINTED) ARCH DERMATOL/ VOL 145 (NO. 9), SEP 2009 WWW.ARCHDERMATOL.COM 1023 ©2009 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/26/2021 rus, stool guiaic, and urine Histoplasma antigen were reported as within the reference range or negative. Serum aspartate aminotransferase and alanine aminotransferase levels were found to be slightly elevated (36 U/L and 39 U/L, respectively, upper limit of normal range, 32 U/L [to convert to microkatals per liter, multiply by 0.0167]). She had evidence of mild anemia with a hemoglobin level of 10.9 g/dL (reference range, 11.5-14.5 g/dL [to convert to grams per liter, multiply by 10.0]), but other blood cell counts were within the reference range. Tests with tissue fungal and mycobacterial stains and cultures were negative. However, skin histologic findings revealed the presence of predominantly intracellular and few extra- cellular microorganisms suspicious for amastigotes (Figure 2). Given the high clinical suspicion for leishmaniasis, additional tissue specimens were obtained and sent with serum to the Laboratory of Parasitic Dis- eases of the Centers for Disease Control and Preven- tion, Atlanta, Georgia. Findings from a tissue smear Figure 1. Leishmania tropica–induced bilateral pretibial ulcerations. demonstrated the presence of amastigotes on light- microscopic examination; tissue culture using Novy, McNeal, and Nicolle media with 10% defibrinated rab- globulinemia.1-3 Viscerotropic leishmaniasis, in contrast bit blood with 15% fetal calf serum was positive, and iso- to visceral leishmaniasis, is a recently described clinical enzyme electrophoresis results yielded L tropica. Serum variant of visceral disease primarily caused by L tropica indirect immunofluorescent antibody (using whole (which traditionally causes cutaneous manifestations) that promastigotes of L donovani) titer for L tropica was el- produces visceral infection but does not necessarily pre- evated at 1:64 (diagnostic titer Ͼ1:16), which, given her sent with the classical manifestations of VL. This is an systemic signs and symptoms, was highly suggestive oligoparasitic syndrome with nonspecific clinical mani- evidence of visceral dissemination of L tropica in this festations caused by the spread of L tropica to the reticu- patient. loendothelial system. This syndrome was initially de- Based on these clinical and laboratory findings, the pa- scribed among 12 US servicemen returning from the tient was diagnosed as having simultaneous cutaneous Persian Gulf War in 1991 with nonspecific symptoms in- and viscerotropic leishmaniasis caused by L tropica. cluding fever, anemia, weight loss, and anorexia.4,5 It is Therapy with a 28-day course of intravenous pentava- notable that none of these originally described cases had lent antimonial compound sodium stibogluconate was concurrent evidence of cutaneous involvement, and sub- provided by the Centers for Disease Control and Preven- sequent reports have also failed to demonstrate this as- tion, Atlanta, Georgia, under an investigational new drug sociation.6,7 Thus, we were interested in reporting a case protocol. A protocol for monitoring potential adverse ef- of concomitant cutaneous and presumptive viscero- fects was performed in our patient, which included a tropic leishmaniasis caused by L tropica in an Afghani weekly electrocardiogram, complete white blood cell refugee who resettled in the United States. count, comprehensive metabolic panel, and pancreatic enzyme measurements. The patient completed treat- REPORT OF A CASE ment successfully. She developed mild hypomagnese- mia, but this promptly corrected with oral supplemen- A 17-year-old Afghani girl who had lived in the United tation. By the end of therapy, the patient had notable States for 4 years presented with a 6-month history of improvement in systemic symptoms, including resolu- bilateral pretibial ulcerations. The patient was born in tion of fatigue, increased appetite, and a weight gain of Kabul, Afghanistan, but soon after was displaced to refu- 1.6 kg. Her splenomegaly and anemia resolved, and her gee camps in Northeastern Pakistan. Subsequent to her liver transaminase levels returned to reference range. At relocation to the United States, she had no history of travel. follow-up 8 months after treatment, the patient’s pre- The patient denied a history of preceding trauma to the tibial ulcerations were reepithelialized, although scar- lower extremities but reported a 9.1-kg weight loss over ring was evident, and she has experienced no signs or the previous 8-month period. In addition, she reported symptoms of relapse. epigastric and hypogastric pain and extreme fatigue af- fecting her daily activities and school performance. Ex- amination revealed a thin girl with nontender spleno- COMMENT megaly. A skin examination revealed 2 ulcerations, one 4ϫ2 cm, the other 2 ϫ 2 cm, on the pretibial aspects of Specific Leishmania organisms are often associated with her lower extremities (Figure 1). Skin biopsy speci- particular clinical presentations such as CL, VL, or ML. mens were obtained for routine histologic tests and tis- However, the relationship between selected species and sue culture. Findings from a chest radiograph and tests clinical syndromes is not always straightforward. In this for antinuclear antibody, human immunodeficiency vi- regard, recent reports have demonstrated the biological

Load more