A 55-year-old woman with human T-cell lymphotropic virus type-1 (HTLV-1)-associated adult T-cell leukemia (ATL) and a history of previously treated Strongyloides stercoralis infection received anti-CD52 monoclonal antibody therapy with alemtuzumab on a clinical trial. After an initial response, she developed ocular involvement by ATL. Alemtuzumab was stopped and high-dose corticosteroid therapy was started to palliate her ocular symptoms. Ten days later, the patient developed diarrhea, vomiting, fever, cough, skin rash, and a deteriorating mental status. She was diagnosed with disseminated S. stercoralis. Corticosteroids were discontinued and the patient received anthelmintic therapy with ivermectin and albendazole with complete clinical recovery.

Keywords:
Adult T-cell leukemia, Alemtuzumab, Corticosteroid, Disseminated Strongyloides, HTLV-1, Human T-cell lymphotropic virus type-1
1.
Tsukasaki K, Hermine O, Bazarbachi A, Ratner L, Ramos JC, Harrington W Jr, O’Mahony D, Janik JE, Bittencourt AL, Taylor GP, Yamaguchi K, Utsunomiya A, Tobinai K, Watanabe T: Definition, prognostic factors, treatment, and response criteria of adult T-cell leukemia-lymphoma: a proposal from an international consensus meeting. J Clin Oncol 2009;27:453–459.
2.
Carvalho EM, Da Fonseca Porto A: Epidemiological and clinical interaction between HTLV-1 and Strongyloides stercoralis. Parasite Immunol 2004;26:487–497.
3.
Brodine SK, Thomas A, Huang R, Harbertson J, Mehta S, Leake J, Nutman T, Moser K, Wolf J, Ramanathan R, Burbelo P, Nou J, Wilkins P, Reed SL: Community-based parasitic screening and treatment of Sudanese refugees: application and assessment of Centers for Disease Control guidelines. Am J Trop Med Hyg 2009;80:425–430.
4.
Dawson-Hahn EE, Greenberg SL, Domachowske JB, Olson BG: Eosinophilia and the seroprevalence of schistosomiasis and strongyloidiasis in newly arrived pediatric refugees: an examination of Centers for Disease Control and Prevention screening guidelines. J Pediatr 2010;156:1016–1018.
5.
Ramanathan R, Nutman T: Strongyloides stercoralis infection in the immunocompromised host. Curr Infect Dis Rep 2008;10:105–110.
6.
Salluh JI, Bozza FA, Pinto TS, Toscano L, Weller PF, Soares M: Cutaneous periumbilical purpura in disseminated strongyloidiasis in cancer patients: a pathognomonic feature of potentially lethal disease? Braz J Infect Dis 2005;9:419–424.
7.
Montes M, Sanchez C, Verdonck K, Lake JE, Gonzalez E, Lopez G, Terashima A, Nolan T, Lewis DE, Gotuzzo E, White AC Jr: Regulatory T-cell expansion in HTLV-1 and strongyloidiasis co-infection is associated with reduced IL-5 responses to Strongyloides stercoralis antigen. PLoS Negl Trop Dis 2009;3:e456.
8.
Nakada K, Yamaguchi K, Furugen S, Nakasone T, Nakasone K, Oshiro Y, Kohakura M, Hinuma Y, Seiki M, Yoshida M, Matutes E, Catovsky D, Ishii T, Takatsuki K: Monoclonal integration of HTLV-1 proviral DNA in patients with strongyloidiasis. Int J Cancer 1987;40:145–148.
9.
Satoh M, Toma H, Sugahara K, Etoh K, Shiroma Y, Kiyuna S, Takara M, Matsuoka M, Yamaguchi K, Nakada K, Fujita K, Kojima S, Hori E, Tanaka Y, Kamihira S, Sato Y, Watanabe T: Involvement of IL-2/IL-2R system activation by parasite antigen in polyclonal expansion of CD4+25+ HTLV-1-infected T cells in human carriers of both HTLV-1 and S. stercoralis. Oncogene 2002;21:2466–2475.
10.
Ratner L, Grant C, Zimmerman B, Fritz J, Weil G, Denes A, Suresh R, Campbell N, Jacobson S, Lairmore M: Effect of treatment of Strongyloides infection on HTLV-1 expression in a patient with adult T-cell leukemia. Am J Hematol 2007;82:929–931.
11.
Agape P, Copin MC, Cavrois M, Panelatti G, Plumelle Y, Ossondo-Landeau M, Quist D, Grossat N, Gosselin B, Fenaux P, Wattel E: Implication of HTLV-I infection, strongyloidiasis, and P53 overexpression in the development, response to treatment, and evolution of non-Hodgkin’s lymphomas in an endemic area (Martinique, French West Indies). J Acquir Immune Defic Syndr Hum Retrovirol 1999;20:394–402.
12.
Porto AF, Santos SB, Muniz AL, Basilio V, Rodrigues W Jr, Neva FA, Dutra WO, Gollob KJ, Jacobson S, Carvalho EM: Helminthic infection down-regulates type 1 immune responses in human T-cell lymphotropic virus type 1 (HTLV-1) carriers and is more prevalent in HTLV-1 carriers than in patients with HTLV-1-associated myelopathy/tropical spastic paraparesis. J Infect Dis 2005;191:612–618.
13.
Thomas MC, Costello SA: Disseminated strongyloidiasis arising from a single dose of dexamethasone before stereotactic radiosurgery. Int J Clin Pract 1998;52:520–521.
14.
Qazilbash MH, Ueno NT, Hosing C, De Lima M, Cortes J, Massaro A, Rivera Z, Deavers M, Adachi JA, Champlin RE: Strongyloidiasis after unrelated nonmyeloablative allogeneic stem cell transplantation. Bone Marrow Transplant 2006;38:393–394.
15.
Keiser PB, Nutman TB: Strongyloides stercoralis in the immunocompromised population. Clin Microbiol Rev 2004;17:208–217.
16.
Genta RM: Dysregulation of strongyloidiasis: a new hypothesis. Clin Microbiol Rev 1992;5:345–355.
17.
Siddiqui AA, Berk SL: Diagnosis of Strongyloides stercoralis infection. Clin Infect Dis 2001;33:1040–1047.
18.
Pelletier LL Jr: Chronic strongyloidiasis in World War II Far East ex-prisoners of war. Am J Trop Med Hyg 1984;33:55–61.
19.
Hirata T, Nakamura H, Kinjo N, Hokama A, Kinjo F, Yamane N, Fujita J: Increased detection rate of Strongyloides stercoralis by repeated stool examinations using the agar plate culture method. Am J Trop Med Hyg 2007;77:683–684.
20.
Goka AK, Rolston DD, Mathan VI, Farthing MJ: Diagnosis of Strongyloides and hookworm infections: comparison of faecal and duodenal fluid microscopy. Trans R Soc Trop Med Hyg 1990;84:829–831.
21.
Loutfy MR, Wilson M, Keystone JS, Kain KC: Serology and eosinophil count in the diagnosis and management of strongyloidiasis in a non-endemic area. Am J Trop Med Hyg 2002;66:749–752.
22.
Schaffel R, Nucci M, Carvalho E, Braga M, Almeida L, Portugal R, Pulcheri W: The value of an immunoenzymatic test (enzyme-linked immunosorbent assay) for the diagnosis of strongyloidiasis in patients immunosuppressed by hematologic malignancies. Am J Trop Med Hyg 2001;65:346–350.
23.
Ramanathan R, Burbelo PD, Groot S, Iadarola MJ, Neva FA, Nutman TB: A luciferase immunoprecipitation systems assay enhances the sensitivity and specificity of diagnosis of Strongyloides stercoralis infection. J Infect Dis 2008;198:444–451.
24.
Gann PH, Neva FA, Gam AA: A randomized trial of single- and two-dose ivermectin versus thiabendazole for treatment of strongyloidiasis. J Infect Dis 1994;169:1076–1079.
25.
Marti H, Haji HJ, Savioli L, Chwaya HM, Mgeni AF, Ameir JS, Hatz C: A comparative trial of a single-dose ivermectin versus three days of albendazole for treatment of Strongyloides stercoralis and other soil-transmitted helminth infections in children. Am J Trop Med Hyg 1996;55:477–481.
26.
Twum-Danso NA: Loa loa encephalopathy temporally related to ivermectin administration reported from onchocerciasis mass treatment programs from 1989 to 2001: implications for the future. Filaria J 2003;2 (suppl 1):S7.
27.
Karunajeewa H, Kelly H, Leslie D, Leydon J, Saykao P, Biggs BA: Parasite-specific IgG response and peripheral blood eosinophil count following albendazole treatment for presumed chronic strongyloidiasis. J Travel Med 2006;13:84–91.
28.
Page WA, Dempsey K, McCarthy JS: Utility of serological follow-up of chronic strongyloidiasis after anthelminthic chemotherapy. Trans R Soc Trop Med Hyg 2006;100:1056–1062.
29.
Turner SA, Maclean JD, Fleckenstein L, Greenaway C: Parenteral administration of ivermectin in a patient with disseminated strongyloidiasis. Am J Trop Med Hyg 2005;73:911–914.
30.
Boken DJ, Leoni PA, Preheim LC: Treatment of Strongyloides stercoralis hyperinfection syndrome with thiabendazole administered per rectum. Clin Infect Dis 1993;16:123–126.
31.
Roxby AC, Gottlieb GS, Limaye AP: Strongyloidiasis in transplant patients. Clin Infect Dis 2009;49:1411–1423.
32.
Santiago M, Leitao B: Prevention of Strongyloides hyperinfection syndrome: a rheumatological point of view. Eur J Intern Med 2009;20:744–748.
33.
Peters L, McCarthy AE, Faught C: Secondary Strongyloides stercoralis prophylaxis in patients with human T-cell lymphotropic virus type 1 infection: report of two cases. Int J Infect Dis 2009;13:e501–e503.
© 2011 S. Karger AG, Basel
2011
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